Education EDF 621 Week 1 Assignment

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Br J Learn Disabil. 2021;49:87–99. wileyonlinelibrary.com/journal/bld  |  87© 2020 John Wiley & Sons Ltd

Received: 11 December 2019  |  Revised: 21 August 2020  |  Accepted: 24 August 2020

DOI: 10.1111/bld.12344

O R I G I N A L A R T I C L E

A multiple methods evaluation of a cognitive behavioural therapy group for people with learning disabilities and anxiety

Rengina Giannaki1,2  | Olivia Hewitt1,2

1Learning Disability Services, Berkshire, UK 2Healthcare NHS Foundation Trust, Reading, UK

Correspondence Rengina Giannaki, Psychology Service, Learning Disabilities, 7-9 Cremyll Road, Reading RG1 8NQ, UK. Email: [email protected]. uk

Accessible summary • Cognitive Behavioural Therapy (CBT) can help individuals with learning disabilities

and anxiety. • A group was run to help people to manage their anxiety and to feel better. • At the end participants and their supporters fed back their experiences of the

group and filled in questionnaires to measure their anxiety and psychological distress.

• The group seems to reduce people's anxiety. • This report adds to a growing body of existing literature demonstrating some ben-

efits of CBT for people with learning disabilities and anxiety.

Abstract Background: Existing studies show mixed results of the effectiveness of CBT for people with learning disabilities and anxiety. Method: A CBT group was run for people with learning disabilities and anxiety, who attended with a supporter. Qualitative interviews were conducted post-group and analysed using thematic analysis. Standardised questionnaires measuring anxiety, psychological distress and overall functioning were administered at pre- and post- group, and at follow-up. Results: Participants’ anxiety decreased overall post-group, which remained largely at follow-up. The group and the supporters’ involvement was helpful. Both supporters and participants recalled coping strategies that were presented in the group. Three out of four participants found attending a group challenging initially. The supporters seem to have specific expectations about the impact of the group. Conclusions: The results of the study are consistent with the existing, growing body of evidence which supports the use of CBT for people with learning disabilities and anxiety.

K E Y W O R D S

clinical psychology, intellectual disability, learning (intellectual) disabilities, mental health, Psychological therapy, stress

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1  | INTRODUC TION

Mental health issues including anxiety are common in people with learning disabilities (Bowring, Painter, & Hastings, 2019; Cooper et al., 2015; NICE, 2016). Reid, Smiley, and Cooper (2011) found that 3.8% of people with a learning disability have an anxiety disorder with generalised anxiety disorder as the most common (1.7%), and then agoraphobia (0.7%). Physical health issues, sensory impairment, specific syndromes and lower cognitive abilities might be associated with the high rates of anxiety in people with learning disabilities (e.g. Austin, Hunter, Gallagher, & Campbell, 2018; Bowring et al., 2019; Dykens, 2003). Anxiety might impact on the quality of everyday life (Bowring et al., 2019) by hindering the social integration (Totsika & Hastings, 2009) and safety of people with learning disabilities. It might be manifested through self-injurious, aggressive or avoidance behaviours (Stavrakaki, 2002).

NICE (2016) indicates that psychological interventions such as cognitive behavioural therapy (CBT) are effective treatments for anxiety within the general population that CBT should be consid- ered to treat depression in people with mild learning disabilities, and that relaxation therapy and graded exposure should be considered to treat anxiety symptoms.

1.1 | The effectiveness of CBT for anxiety in people with learning disabilities

The existing literature shows mixed results for CBT interventions on anxiety in people with learning disabilities. Dagnan's, Jackson, and Eastlake (2018) systematic review reported positive outcomes of CBT interventions on anxiety for people with learning disabilities, although much of the current literature consists of single case re- ports. Unwin, Tsimopoulou, Kroese, and Azmi (2016) systematically reviewed the literature around the effectiveness of CBT and con- cluded that there is equivocal evidence for improving anxiety with some uncontrolled studies demonstrating some positive results, and with the qualitative data consistently reflecting a positive percep- tion of the treatment. Both authors concluded that the area needs further research.

Some studies show limited or no effectiveness of CBT on anxi- ety. Hassiotis et al. (2013) conducted a randomised control trial to study the effectiveness of a 16-week manualised individual CBT on anxiety and depression. Sixteen adults with mild to moderate learn- ing disabilities were randomly allocated in the CBT group and six- teen in the control group. No statistically significant difference was found. However, the approach of intervention was generalised and not anxiety-focused (Unwin et al., 2016).

Other studies indicate a more promising impact of CBT on anxi- ety. Ghafoori, Ratanasiripong, and Holladay (2010) reported a signif- icant reduction in anxiety, following a 9-week generalised approach of CBT group which included eight participants, but that phobic anx- iety was not reduced. Quantitative analysis showed that the anxi- ety remained roughly decreased at 4-month follow-up but without

statistical significance. However, the participants were young stu- dents which limits the generalisability.

Lindsay (1999) used individual Beck's CBT adapted to anxiety and found that the fifteen participants’ anxiety scores decreased by 35%, which persisted at follow-up. However, the study comprised a brief case report and presented little methodological information (Unwin et al., 2016).

Lindsay et al. (2015) conducted a matched control trial employing a quasi-experimental design. The CBT group involved twelve adults with mild learning disabilities with their supporters. The author developed a CBT manual containing a generalised approach targeting mixed presen- tations, anxiety, depression and anger. Significant decrease in anxiety was found post-group which remained at 6-month follow-up. However, no between group treatment effects on anxiety were reported.

Douglass, Palmer, and O'Connor (2007) studied the effective- ness of a CBT group for anxiety. Outcome measures demonstrated that after a 12-week intervention, two out of six participants noted a significant reduction in anxiety. All group members developed cop- ing strategies. Four participants reported decreased anxiety and all supporters a better understanding of anxiety. The study used both quantitative and qualitative data, but did not present quotes sup- porting the qualitative analysis.

In Marwood and Hewitt’s (2012) study, quantitative data sug- gested that two out of eight participants with mild learning disabil- ities noted a significant decrease of anxiety after a 6-week CBT group. Qualitative data analysis reported positive experiences of attending the group and of supporters’ inclusion. However, the num- ber of participants who noted a decrease of anxiety remains small compared to the total number of participants, though each partici- pant reported finding the group beneficial.

Given the limited existing literature and the ambiguity of results, an evaluation of a CBT intervention seems to be appropriate in de- termining its efficacy on anxiety. According to NICE (2016), further research is needed to evaluate the effectiveness of CBT in people with learning disabilities and anxiety. Due to the limited qualitative data and the limited opportunities that people with learning dis- abilities have to express their views on interventions, a qualitative methodology in addition to a quantitative one could enrich the data and enable the voices of people with learning disabilities to be heard allowing a deeper understanding of participants’ experience in the group. It will also increase insights into helpful components of the intervention and into the process of change (MacMahon et al., 2015).

This study aims to evaluate the effectiveness of a CBT group for adults with learning disabilities who present with anxiety symptoms impacting on their everyday life to the extent that psychological input is sought. A secondary objective is to investigate if the inter- vention will help participants to feel better by reducing their psycho- logical distress.

2  | METHODOLOGY

A service evaluation was carried out using multiple methods.

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2.1 | Participants

The participants who attended the group consisted of four indi- viduals with mild learning disabilities and anxiety symptoms (see Table 1 for demographic information). All of them were presenting with symptoms related to generalised anxiety and specific fears. Each participant was asked to attend the group with a supporter, either family member either carer, who could commit to the group. Previous research reported benefits of supporters’ inclusion by generalising the learned skills in other areas of participants’ life and maintaining long-term treatment effects (e.g. Crossland, Hewitt, & Walden, 2017).

Participants were referred to the psychological service for peo- ple with learning disabilities within the NHS Trust and were assessed for suitability for the group. Inclusion criteria were being over the age of 18 years, presenting with anxiety symptoms, having mild to moderate learning disabilities, being willing to participate in the group, and a supporter's inclusion. No formal, psychiatric diagno- sis of anxiety was required. Exclusion criteria were receiving other psychological therapy during the group or having severe learning disabilities.

All group members gave informed consent both to participate in the group and in its evaluation. They were informed about the ano- nymity of the information they provided. The project was reviewed and registered with the research department of the NHS Trust, which approved it as a service evaluation without warranting review by the research ethics committee.

2.2 | Group content

The group content consisted of basic CBT concepts based on Beck’s (1987) model. The material was developed in the service (see Table 2 for outline of group sessions). Cognitive and behavioural compo- nents were introduced in the group as guided by previous research (e.g. Dagnan et al., 2018; Douglass et al., 2007 for review; NICE Guideline 54, 2016).

Cognitive behavioural therapy is often adapted for use with peo- ple with learning disabilities. Adaptations include using visual aids and prompts, using diary sheets requiring only tick box responses, shorter sessions with slower pace, using concrete examples, re- peating information (Haddock & Jones, 2006). Such adaptions were

included in the present group. Previous research suggests includ- ing supporters improves outcomes (e.g. Douglass et al., 2007). Therefore, each participant was encouraged to bring a supporter to the group to support generalising new skills.

The programme consisted of a 7-week CBT group targeting anx- iety and one follow-up session taking part one month after the last of the 7-week sessions. Two trainee clinical psychologists and two assistant psychologists facilitated the group receiving weekly super- vision by a clinical psychologist.

2.3 | Measures

Three standardised measures were administered by a trainee clini- cal psychologist and an assistant psychologist to the individuals with learning disabilities who attended the group at three time points. A range of measures comprising specific and generic scales were em- ployed in support of the aims of the study. All three detect changes of both anxiety and overall mental health as interventions in an area can lead to changes in several areas of functioning (Roy, Matthews, Clifford, Fowler, & Martin, 2002; Unwin et al., 2016). These meas- ures were chosen for their psychometric properties and were used in previous research (e.g. Douglass et al., 2007). One additional ques- tionnaire, the PTOS-ID, was administered to participants’ supporters at three time points to capture supporters’ perceptions of partici- pants’ level of distress and to identify any discrepancies among par- ticipants’ and supporters’ perception.

The Glasgow Anxiety Scale for people with an Intellectual Disability (GAS-ID; Mindham & Espie, 2003) is a measure designed for people with mild learning disabilities. It has good reliability and internal consistency. The 27-item self-rating scale evaluates the level of anxiety and comprises of cognitive, behavioural and somatic symptoms that the person experiences over the past week. The au- thors suggest a cut-off score of 13 as a threshold value to identify a possible anxiety disorder. Higher score shows higher degree of anxiety. The three-option response format, “never,” “sometimes,” “always”, makes the measure sensitive to changes over time.

The Health of the Nation Brief Outcome Measure for People with Learning Disabilities and Mental Health Needs (HoNOS-LD; Roy et al., 2002) rates the mental health needs and global function- ing for people with all levels of learning disabilities. The 18-item in- strument has good reliability and validity, and can measure changes

TA B L E 1   Demographic data

Pseudonym Age Ethnicity Gender Living situation Diagnosis Supporter's pseudonym and relationship

Alice 21 British-White Female Family home Mild LD, anxiety Sylvia, mother

Colin 56 British-White Male Independent living with support staff

Mild LD psychosis, anxiety

Ivor, paid support worker

Mary 48 British-White Female Supported accommodation

Mild LD, anxiety Jacob, partner

Sally 55 British-White Female Family home Mild LD, anxiety Erica, mother

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over time. The clinician rates each item over the past four weeks according to the level of severity of the problem scoring from 0 to 4. Higher level of wellbeing is indicated by lower total score. The measure can detect changes over time.

The Psychological Therapy Outcome Scale-Intellectual Disabilities (PTOS-ID; Vlissides, Beail, Jackson, Williams, & Golding, 2017) is a 30-item self-rated scale designed to measure anxiety, anger, depres- sion, interpersonal wellbeing and psychological wellbeing in people with mild to moderate learning disabilities. Each item is rated on a 4-point Likert scale anchored by “not at all” to “a lot.” The measure as- sesses two dimensions over the past week: the index of psychological distress and the index of wellbeing. The psychological distress score equals the sum of anxiety, depression and anger scores. Higher score of the psychological distress index indicates higher level of distress. The wellbeing score equals the sum of interpersonal wellbeing and psychological wellbeing. A parallel PTOS-ID form can be completed by supporters and captures supporters’ perception about the distress and wellbeing of the person with a learning disabilities. The measure has high internal consistency of the psychological distress index (α = 0.85) and high reliability of the wellbeing index (α = 0.81). Full evaluation of its psychometric properties has yet to be carried out.

2.4 | Qualitative method

Qualitative data were derived from semi-structured interviews which were conducted post-group in order to obtain group members’ experiences of the group. The interviews were conducted at par- ticipants’ homes, separately for each participant and each supporter. This allowed all group members’ voices to be heard independently and to compare participants’ and their supporters’ experience. A trainee clinical psychologist and two assistant psychologists carried

out the interviews after receiving participants’ and supporters’ writ- ten, informed consent.

The interviews included questions related to the aims of the study: participants’ experience of the group, their interaction with others, group content, what participants learned, helpful and un- helpful things from the group. An interview schedule contained questions which were used in similar studies. A pilot interview took place among the interviewers who provided with feedback to each other. Alongside a discussion with the supervisor ensured the appro- priateness of the questions and of the interview process. The ques- tions were open-ended with prompts, and they were used flexibly as a guide allowing a natural flow of conversation.

The interviews lasted between 13 and 30 min. They were audio recorded using digital voice recorder as explained in the information provided. The recordings were transcribed verbatim by the same interviewers respectively. Identifying information in the transcripts was anonymised.

The transcripts were analysed by the first author using the- matic analysis. Thematic analysis is a method for identifying, an- alysing and reporting patterns (themes) within data. It organises, describes the data and interprets various aspects of the research topic. Six stages of thematic analysis were used in the current study (Braun & Clarke, 2006). The first author was familiarised initially with the data by listening to the audio recordings and reading the transcripts repeatedly; the author created initial codes involving collating and coding quotes (coding); the author looked for similar themes in the transcript and grouped them based on the similarity (searching for themes); the author reflected on the themes, discussed them in the supervision and edited them (re- viewing themes). The fifth stage of “defining and naming themes” was achieved by creating broader superordinate themes of similar themes. The qualitative data were divided into participants’ data and into supporters’ data to ensure that participants’ voice is heard and to achieve a broader understanding of the group members’ experience. The sixth stage included the writing of this report, reflecting, receiving feedback from the supervisor and making choices about the quotes presented.

To ensure credibility, detailed notes were undertaken for all the processes by the first author throughout the study. Regular supervi- sion of the analysis also provided by OH, a clinical psychologist with experience in conducting qualitative research. To ensure validity and trustworthiness, the first author was repeating detailed readings of the verbatim transcript. The first author's role as co-facilitator of the group allowed an early familiarity with the concepts of the group and the establishment of a relationship of trust with the interviewees ensuring validity as well (Shenton, 2004).

2.5 | Attendance and missing data

All four participants completed the group. All measures were completed for all participants at all three time points (pre-, post- intervention and at follow-up). Three out of four participants

TA B L E 2   Group session outline

Week Topic

1 Introduction, creating a safe environment, overview of the programme, supporters’ role

Introducing what anxiety means, anxiety provoking situations, helpful coping skills

2 Identifying emotions. hometask: recording emotions

3 Identifying unhelpful thoughts and their impact on emotions and actions. Setting optional hometask: breathing relaxation

Three weeks Christmas break

4 Recap of first three sessions

5 Identifying body reactions of anxiety

6 Identifying actions. Five senses self-soothing activity

One week break

7 Linking emotions, thoughts and behaviours

8 One month follow-up. Recap of the programme. Completing measures

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completed the measures at 1-month follow-up after the end of the group. Due to Colin's physical health issues, the administration of his follow-up measures took place 2-month post-group. One participant, Mary, joined the group from the fourth group session and she was not administered the HoNOS-LD at pre-group. Alice's interview was not recorded due to technological issues thus her interview is missing.

3  | RESULTS

3.1 | Quantitative results

The group mean and the standard deviation for the three measures are displayed in Table 3. All participants’ pre-group mean score of anxiety for all three measures decreased by nearly half following the intervention. The group mean of anxiety for GAS was 25.25 at intake and 13 post-group. The pre-group mean of distress for PTOS-ID was 25.25 and 13.75 post-group. The mean for three out of four participants on HoNOS-LD was 16.33 initially and 8.25 post-group. The reduced scores remained at follow-up (Figure 1). The group mean of participants’ distress on PTOS-ID as perceived by supporters was 19.5 at intake and became 5.25 at post-group,

which was not maintained at follow-up, where it reverted to base- line levels (Table 3).

Due to the small number of participants, a Reliable Change Analysis (RCI) was carried out for the three time points. This analysis compares each participant's individual scores over time to see if the changes are significant (Table 3).

3.1.1 | Alice

Alice's post-group anxiety score on GAS decreased (RCI = −2.20, p < .05) which remained at follow-up (RCI = −1.69, p < .05) compared to pre-group. Alice's post-group score on HoNOS-LD decreased meaningfully (RCI = −1.88, p < .05), which persisted at follow-up de- spite a slight increase (RCI = −1.61, p < .05).

The Figure 2 shows a significant reduction of distress post- group both on PTOS-ID for Alice (RCI = −2.24, p < .05) and on PTOS-ID completed by her supporter (RCI = −1.36, p < .05). Alice's score of distress on PTOS-ID at follow-up (RCI = −0.46, p < .05) remained at low level though it was slightly equivalent to the post- group score. Alice maintained a meaningful low score on support- er's PTOS-ID at follow-up (RCI = −0.24, p < .05) despite a slight increase.

Measure/Time Alice Colin Mary Sally Mean score

Standard deviation

GAS pre 23 21 23 34 25.25 5.90

GAS post 10 10 18 14 13 3.82

GAS F/U 13 12 15 12 13 1.41

GAS RCI pre-post −2.20* −1.86* −0.84* −3.38*

HoNOS-LD pre 29 8 – 12 16.33 11.15

HoNOS-LD post 8 0 2 23 8.25 10.40

HoNOS-LD F/U 11 14 3 16 11 5.75

HoNOS-LD RCI pre-post

−1.88* −0.71* – 0.98

PTOS Distress pre

41 21 17 22 25.25 10.71

PTOS Distress post

17 18 10 10 13.75 4.34

PTOS Distress F/U

36 22 12 8 14 4.96

PTOS Distress RCI pre-post

−2.24* −0.28* −0.27* −0.47*

Supporter's PTOS Distress pre

20 22 12 8 19.5 12.47

Supporter's PTOS Distress post

3 12 0 6 5.25 5.12

Supporter's PTOS Distress F/U

17 16 30 11 18.5 8.1

Note.: Higher scores indicate increased impairment. Abbreviations: F/U, follow-up; Post, post-intervention; Pre, pre-intervention; RCI, reliable change index. *Statistically significant at α = 0.05.

TA B L E 3   Participants’ pre-post reliable change index (RCI) scores

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3.1.2 | Colin

A RCI analysis demonstrated that Colin's GAS score lowered signifi- cantly post-group (RCI = −1.86, p < .05) and at follow-up (RCI = −1.52, p < .05). Colin's score on HoNOS-LD decreased immediately post- group scoring 0 (RCI = −0.71, p < .05) indicating a meaningful im- provement in his global functioning post-group. Colin's post-group reliable change on HoNOS-LD was not maintained at follow-up showing a decrease of his global functioning (RCI = 0.53) compared to pre-group. This might be attributed to Colin receiving fewer inter- personal interactions post-group or to a physical condition.

A RCI analysis showed a meaningful reduction of distress on the index of distress on PTOS-ID for Colin post-group (RCI = −0.28, p < .05) which was not maintained at follow-up (RCI = 0.09). Colin's score of distress on supporter's PTOS-ID at follow-up was reduced significantly (RCI = −0.80, p < .05) post-group despite a slight in- crease of distress at follow-up compared to post-group (Figure 3).

3.1.3 | Mary

Mary noted a reliable reduction of her anxiety level (RCI = −0.84, p < .05) on GAS at post-group, which decreased further at follow- up (RCI = −1.35, p < .05). Mary did not fill in the HoNOS-LD before the group thus it was not possible to track any change of her overall functioning over time.

Mary's score of distress for PTOS-ID demonstrated reliable change and her level of distress decreased post-group (RCI = −0.27, p < .05) and remained at a lower level at follow-up (RCI = −0.46, p < .05) despite a slight increase. Mary's score on supporter's PTOS-ID noted a reliable change post-group (RCI = −0.96, p < .05) indicating a significant decrease of her distress. Mary showed a high increase of distress on supporter's PTOS-ID at follow-up (RCI = 1.44) which was higher than the pre-group score (Figure 4). This might be because the measure was administered to two different persons. Alongside her partner reported in the interview no improvement for Mary. This might be because Mary joined the group after the other participants and she did not apply the coping strategies outside the group.

3.1.4 | Sally

From the RCI emerged that Sally's anxiety score for GAS de- creased meaningfully post-group (RCI = −3.38, p < .05) which remained almost equivalent at 1-month follow-up (RCI = −3.22, p < .05). Sally's score on HoNOS-LD was increased post-group (RCI = 0.98) indicating an increased impairment in her global func- tioning. There was a slight improvement in her functioning at fol- low-up (RCI = 0.35) compared to the post-group which remained lower than the pre-group (Figure 5). Sally's post-group score on HoNOS-LD was increased showing decreased functioning, possi- bly due to a physical condition that Sally developed whilst attend- ing the group.

Sally's score on PTOS-ID noted a reliable decrease (RCI = −1.12, p < .05) immediately after the intervention showing an improvement of her distress. The post-group reliable change remained at follow-up (RCI = −1.30, p < .05) showing a slight higher level of improvement compared to the intake. Sally's score for supporter's PTOS-ID noted a reliable reduction of distress (RCI = −0.16, p < .05) at post-group but was slightly increased at follow-up (RCI = 0.24) compared to pre- group. This might be due to the ending of the group, which was a positive experience for Sally.

Clinical significance was used to examine the pre- and post- group changes for each participant. Clinical significance is

F I G U R E 1   Participants’ changes for HoNOS-LD, GAS, PTOS-ID index of distress and supporter’s (S/R) PTOS-ID index of distress at pre-, post-group and follow-up. Higher scores on all measures indicate an increase of impairment and vice versa

0 5

10 15 20 25 30

Group's mean scores over time

F I G U R E 2   The graph displays Alice's scores at pre, post and follow-up intervention for the measures GAS, HoNOS-LD, index of distress on PTOS-ID and index of distress on supporters’ (S/R) PTOS-ID. Higher scores indicate increased level of impairment

0 10 20 30 40 50

Alice's scores over time

F I G U R E 3   The graph displays Colin's scores at pre-, post- and follow-up intervention for the measures GAS, HoNOS-LD, index of distress on PTOS-ID and index of distress on supporters’ (S/R) PTOS-ID. Higher scores indicate increased level of impairment

0 5

10 15 20 25

Colin's scores over time

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routinely defined as returning to normal functioning, and occurs when post-intervention scores are more than two standard de- viations above or below the mean score of the pre-intervention sample (Jacobson, Roberts, Berns, & McGlinchey, 1999). Alice's and Colin's post-group improvement on all measures were clini- cally significant. This is supported for Alice by subjective reports. Mary's and Sally's improvement on two measures were clinically significant, which is in line with subjective reports for Sally; how- ever, this is contradicted for Mary by her supporter's subjective reports.

3.2 | Qualitative results

The quotes below indicate text taken directly from the transcripts. The number in brackets indicates the line location within the data. The “P” refers to participants’ and “S” to supporters’ quote.

3.2.1 | Results from participants

Four superordinate themes were identified from the participants’ interviews (Table 4).

Superordinate theme 1: How things were before the group The first superordinate theme described participants’ pre-existing issues and helpful coping skills which were used before starting the group.

All participants identified emotional difficulties, such as fears, frustration and family problems. Mary and Colin expressed concerns around medical appointments. Sally said:

You get headaches. Bad headaches sometimes. (P, 133)

Colin described:

Years ago I used to be a little bit scared of dogs… If we go to the doctor and somebody does not explain me the reason that bothers me.

(P, 52)

All participants described how they coped with anxiety before the group. Mary engaged in helpful activities such as reading and writing. Two participants used physical activity to help them to relax. Collin said:

I use my exercise bike… sometimes I do jogging. And I count up to ten. And I jump up until twenty… it gets out my stress.

(P, 88)

Superordinate theme 2: Participants’ perceived positive and negative aspects of the group This theme considered participants’ positive experience in the group, how they benefited from the group and things they did not like about the group.

All participants held a generally positive view of the group. Two participants enjoyed the group content and activities. Sally found the group “interesting” and said:

it was all good. That was a nice change. I enjoyed it. It was good to do things. I had a nice time.

(P, 171)

Most participants learned helpful coping strategies to manage anx- iety. Two participants found it helpful to talk about their problems in the group. Mary said that she learned:

to talk about my anxiety, the appointment at the hos- pital that I didn't like.. I have to go for that… to talk about to my sister, and feel better…talk about your problems, anxiety it helped me too.

(P, 8)

F I G U R E 4   The graph displays Mary's scores at pre-, post- intervention and follow-up for GAS, HoNOS-LD, index of distress on PTOS-ID and on supporters’ (S/R) PTOS-ID. Higher scores indicate increased level of impairment. Mary's HoNOS score at pre- group is missing

0 5

10 15 20 25 30 35

Mary's scores over time

F I G U R E 5   The graph displays Sally's scores at pre-, post- and follow-up intervention for GAS, HoNOS-LD, index of distress on PTOS-ID and on supporters’ (S/R) PTOS-ID. Higher scores indicate increased level of impairment

0 10 20 30 40

Sally's scores over time

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Two participants described difficulties they experienced in the group. One participant found the room “dark” and the venue too small. Another participant found it difficult initially to be in a group with others.

Superordinate theme 3: Participants’ perceived positive interactions with other group members This theme incorporates participants’ positive experience of inter- acting with other participants, supporters and facilitators.

Sally and Colin enjoyed talking to other group members. Colin described:

I‘ve enjoyed having a chat. And, I‘ve enjoyed explain- ing about myself. I really enjoyed the group.

(P, 16)

Sally liked learning from others and said that it was “nice” to “talk to your friends.” She liked the communication among group members:

[liked] listening and hearing things. Stick to other peo- ple's conversations. Taking in turns. Well done.

(P, 11)

All participants expressed their appreciation about the facilitators describing them as “helpful” and “nice.” For example, Colin said: “I‘ve enjoyed having a chat with them” (P, 38). One participant, Colin, ex- pressed a positive view of his supporter's inclusion: “I really enjoyed it with Ivor in group.” (P, 234).

Superordinate theme 4: Group content This theme reflects topics that were presented in the group, activities that were practised in the group to deal with anxiety and the hometask.

All participants recalled some of the group topics such as feel- ings, behaviours and bodily sensations. Sally recalled that “we talked about heart” and about:

the very happy face on the board. That‘s a good one… It is funny that.

(P, 53)

She also recalled an activity involving writing anxiety provoking thoughts on a picture of a head. She liked the “nice big circle eyes…you could draw it.” (P, 90).

Two participants talked about their positive contribution to the group being a model for others as they presented their pre-existing helpful coping strategies. For example, Sally shared with others what she described as “stretching”:

Looking up to the ceiling, and then looking down. That's good. You have to be careful with your neck…I did it in the anxiety group. It is good Sally because you move and your little fingers as well.

(P, 145)

All participants completed the hometask at least once with their supporters’ help. Most liked the hometask. Sally found it “quite good” although Mary said that she did not find it helpful.

3.2.2 | Results from supporters

Five superordinate themes were identified from the supporters’ in- terviews (Table 5).

Superordinate theme 1: How things were before the group The first theme captures supporters’ account of both participants’ and their own emotional issues, coping skills, positive things in eve- ryday life before the group and expectations about the group.

Most supporters said that the participants were encountering emotional difficulties in their everyday life before starting the group. Jacob said:

Superordinate theme Subordinate theme Endorsed by

1. How things were before the group

Participants’ pre-existing emotional difficulties

Participants’ pre-existing coping skills

Colin, Mary, Sally

2. Perceived positive and negative aspects of the group

Participants’ positive experience in the group

How the group helped participants Participants’ difficulties in the group

Colin, Mary, Sally

3. Participants’ perceived positive interactions with other group members

Participants’ positive experience of interacting with other group members

Colin, Mary, Sally

4. Group content Topics and activities in the group Participants’ presentation of coping

skills Hometask

Colin, Mary, Sally

TA B L E 4   Summary of superordinate and subordinate themes for participants

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Mary gets worried sometimes. She wants me to go always out and get her stuff… (S, 13). When she does things, when she goes somewhere, she always likes somebody with her.

(S, 213)

Sylvia talked about Alice's social withdrawal and Erica about Sally's fears. Most supporters described their own worries. Erica expressed her initial hesitation to attend the group:

I wasn't very keen on going to the group first of all. I couldn't believe that [Sally] would change.

(S, 5)

Superordinate theme 2: What supporters found helpful about the group This theme captures group aspects which supporters perceived as helpful for the participants and themselves, such as the group envi- ronment and content, the hometask, the interactions among group members, practical things.

All supporters described the group as “relaxed,” “interesting” and “friendly” reporting an overall positive experience. For example,

Erica felt “happy”. They appreciated the safe and non-judgemental environment where people felt free to express their views and needs.

All supporters recalled topics from the group and most of them endorsed the group content reporting its positive impact on them- selves, despite materials being targeted to participants. They found the strategies to deal with anxiety and the activities that were prac- tised in the group helpful and enjoyable. Introducing coping strate- gies from the first sessions helped Alice to manage her anxiety as she became familiar with the group, her mother said. Erica found everything was helpful:

it did really sink in all those things for her [Sally]. And she does talk about those things, when she gets home.

(S, 6)

Half of supporters found the hometask which was completed by all participants at least once as helpful. Sylvia recognised that this activity is a “massive help.” Jacob found it “good” and that it “helps me under- stand the feelings what is like.”

Most supporters found the social aspect helpful. Ivor empha- sised the opportunity of making friendships and the usefulness of

Superordinate theme Subordinate theme Endorsed by

1. How things were before the group

Participants’ and supporters’ emotional difficulties in life

Participants’ and supporters’ coping strategies and positive things in life

Supporter's initial hesitation to attend the group

Erica, Jacob, Ivor, Sylvia

2. What supporters found helpful about the group

Supporters’ perceived positive aspects of the group

Supporters’ perceived positive group environment Supporters’ perceived positive group topics and

coping strategies Supporters’ perception about the hometask Social aspects of the group are helpful Others as a source of learning Practical aspects of the group are not an issue

Erica, Jacob, Ivor, Sylvia

3. Supporters’ perceived negative aspects of the group

Participant's initial expectation about the group is not met

Supporters’ perceived negative social aspects of the group

Supporters’ perception about participants’ difficulties in the group and after the termination of the group

Supporter's negative experience about the hometask

Supporter's negative experience about practical aspects of the group

Erica, Jacob, Ivor, Sylvia

4. Supporters’ perceived positive impact of the group on participants and themselves

Supporters’ perceived positive impact of the group on participants’ emotions in the group

Supporters perceive a positive impact of the group on participants which lasts after its termination

Erica, Jacob, Ivor, Sylvia

5. Looking to the future Supporters’ constructive feedback about the group Participant's care plan after the group Supporters’ hopes for the future

Erica, Sylvia, Ivor

TA B L E 5   Summary of themes from supporters’ interviews

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discussions. Most supporters perceived their role as active and helpful. All supporters expressed their appreciation for the facilitators. Sylvia said:

the way you did it passing the bottle and that that helped that helped Alice as well. Alice came out of her shell in the last week… which was good to see what a difference.

(S, 112)

Supporters felt participants benefited from learning through oth- ers’ experiences and skills. Colin got help hearing that “other people have anxieties,” Ivor said. Erica said: “listening to those she [Sally] got confidence from those how to answer questions.” (S, 24). Sylvia said: “it was good to get different perspectives.”

Superordinate theme 3: Supporters’ perceived negative aspects of the group Supporters described negative aspects of the group and members’ difficulties in the group and after the end.

Sylvia felt the facilitators should have been more aware of par- ticipants’ needs before starting the group. Talking about the range of participants’ age Sylvia said:

She [Alice] was running off, she was getting very frus- trated and anxious, but I think that was because Alice was expecting it to be her age range and it wasn't, it was her and two 50 plus year olds and she found that difficult…That was a shame because there was no- body that she could become friends with afterwards.

(S, 7)

Jacob expressed his preference for an individual intervention. Half of supporters expressed their dissatisfaction at one participant's dominance of the conversation. Two participants, Sally and Mary, felt uncertainty about what to say. With regards to the hometask, Erica felt initially “a little bit muddled” on “how to fill those spaces [on the worksheets].” Parking was a “big issue” for one supporter. Jacob said that Mary has not used the coping strategies from the group and that:

She [Mary] still gets worries and stress. Because she came back and then in few days, she is worrying about things again.

(S, 230)

Superordinate theme 4: Supporters’ perceived positive impact of the group on participants and themselves The theme captures supporters’ perception about the positive out- come of the group on participants’ feelings and behaviours in the group and after the end of the group.

Half of supporters talked about participants’ positive feelings and changes in the group over time. Sylvia noticed that Alice became “relaxed” and that she had a “massive difference” at the end with

“fully engagement.” Erica mentioned Sally “found so happy to speak to other people.”

Ivor noticed two participants’ progress: “in a very short space of time, she [Alice] grew to trust the group and enjoyed interacting. I thought that was really impressive.” (S, 10).

Supporters described participants’ feelings and behaviours which lasted after the termination of the group. Erica noticed Sally's “tremendous difference” as she “talks more,” “won't worry about anything” and “feels happier.”

But the happiness she got at the group was really, helped her down there [day centre] really. Because she kept saying I am a nice person, I am strong, I am gonna be all the things that you said at the group, she is taking that on board and repeats that.

(Erica, 11)

Sylvia commented that the “healing process” started early and that the group helped Alice:

definitely one hundred percent… she doesn't get as anxious anymore, she's now started going out more with her friends. She went to the pub last night with a few friends.

(S, 234)

Superordinate theme 5: Looking to the future This theme incorporated suggestions for future groups, plans for on- going psychological support and hopes for the future. Sylvia would prefer the group content to be tailored to each participant's needs:

what’ s the requirement for the individual, what is it that they need from the group, you need to find that out.

(S, 179)

Erica expressed her hope that Sally's progress will last in long-term:

I am hoping now, that we can go forward back to how it used to be one time, when she [Sally] was going to shopping.

(S, 28)

4  | DISCUSSION

This study aimed to evaluate the effectiveness of a 7-week CBT group intervention for adults with learning disabilities and anxiety using a multiple methods design, combining standardised measures with qualitative analysis.

Quantitative results show that the CBT group had an overall positive impact on most participants’ anxiety, distress and global functioning, which was maintained for most at follow-up. This study

     |  97GIANNAKI ANd HEWITT

replicated findings that CBT might be helpful for people with learn- ing disabilities and anxiety (Dagnan et al., 2018).

Participants’ mean and individual GAS scores decreased post- group, which remained at follow-up as shown previously (e.g. Lindsay et al., 2015). Participants’ HoNOS-LD scores improved post-group in line with Marwood and Hewitt's study (2012).

All participants’ post-group level of distress decreased, which remained at follow-up despite a slight higher score of distress for most of them, as supported by Crossland et al.'s study (2017). The post-group level of distress on supporter's PTOS-ID for all participants lowered, and the level of distress at follow-up re- mained at low level for three out of four participants despite a slight increase.

The scores of measures are fairly consistent with the qualitative results. The qualitative analysis showed that the group intervention was largely a positive experience for participants who reported get- ting help from the group and from interacting with others, possibly through normalising their own experiences (Marwood & Hewitt, 2012). Based on supporters’ narratives, half participants reported a decrease of anxiety and increase of wellbeing which is supported elsewhere (e.g. Bouvet & Coulet, 2015).

The qualitative data showed that most supporters worried about participants’ wellbeing, as reported elsewhere (e.g. Mansell & Wilson, 2010). Supporters helped participants to manage their initial worries, generalise skills outside the group (e.g. Douglass et al., 2007) and engage with the group (Willner, 2006). Supporters benefited from learning coping strategies for themselves as well, as shown previously (Crossland et al., 2017).

Regarding the group content, participants recalled some anx- iety-related feelings, behaviours and body reactions. Most par- ticipants recalled the skills practised in the group (mindfulness and relaxation skills), in line with similar studies (e.g. Bouvet & Coulet, 2015). Participants presented their own pre-existing coping skills in the group. Using visual aids appears to have aided recollec- tion of the content (Haddock & Jones, 2006).

Some participants recalled anxiety-related situations and be- haviours. This is in line with Tsimopoulou, Stenfert Kroese, Unwin, Azmi, and Jones (2018) study demonstrating that people with a learning disability learned to link activating situations to emo- tions. None of the participants discussed the impact of unhelpful thoughts on anxiety, which is a core concept of cognitive therapy (Beck, 1987), which was found elsewhere (Vereenooghe, Reynolds, Gega, & Langdon, 2015) suggesting that people with learning dis- abilities did not learn to link thoughts to emotions. This might reflect participants’ difficulty in perceiving or recalling abstract concepts. Problems recalling theoretical elements are replicated by other re- search (e.g. Hewitt, Atkisnon-Jones, Gregory, & Hollyman, 2019).

4.1 | Clinical implications

The CBT group had an overall positive impact on participants. The quantitative analysis showed improvement whilst the qualitative

analysis showed that the group was largely beneficial for partici- pants. Current findings have implications for services working with people with learning disabilities.

From the qualitative data, it seems that group aspects that helped achieving positive results are the visual presentation of con- cepts, the presentation of basic CBT concepts such as situations, behaviours, body reactions that are related to anxiety, the practice of skills in and outside the group and modelling participants’ own pre-existing skills to others, all enhanced learning. An environment promoting safety, the social interaction and supporters’ involvement helped too.

Exploring individuals with learning disabilities unhelpful thoughts eliciting anxiety, participants’ and supporters’ initial expectations about the intervention might be helpfully integrated in the assessment session. Setting any unrealistic initial expectations might be of benefit before starting the group. Given the supporters’ self-reported anxiety, additional sessions could focus on supporters’ anxiety, which might enhance participants’ progress over time. Encouraging participants’ regular attendance could enhance participants’ learning.

4.2 | Research implications

This study suggests promising results around the benefits of a CBT group for people with learning disabilities, adding to the growing lit- erature. Use of multiple methods allowed tracking the improvement with regards to anxiety and psychological distress. Standardised measures were used to ensure validity. A qualitative results both from participants and their supporters allowed in depth exploration of what aspects of the group were most helpful for participants. A fol- low-up evaluation helped to identify if the gains remained over time.

However, the results following the intervention should be inter- preted with caution. Supporters reported external factors that may have impacted on measures of anxiety. The first author held the role of group facilitator and interviewer holding possibly biases around the results. A lack of control group limits the reliability of the study. The small sample and large standard error limit the generalisability of the results. The specific cognitive changes linked to participants’ anxiety have not been captured over time. Administering outcomes measures at three time points to the same person with consistency could reduce any errors. Future research should be more rigorous, using more robust methodologies with larger samples of people to ascertain which aspects of CBT are effective and the generalisability of the results.

ACKNOWLEDG EMENTS R.G. would like to thank Iman Hassan, trainee clinical psychologist, Katie Ash and Nerissa Grant, assistant psychologists for their help throughout the process.

DATA AVAIL ABILIT Y S TATEMENT The data that support the findings of this study are available on re- quest from the corresponding author, Rengina Giannaki. The data

98  |     GIANNAKI ANd HEWITT

are not publicly available due to their containing information that could compromise the privacy of research participants.

ORCID Rengina Giannaki https://orcid.org/0000-0002-1807-1347 Olivia Hewitt https://orcid.org/0000-0002-6393-2388

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How to cite this article: Giannaki R, Hewitt O. A multiple methods evaluation of a cognitive behavioural therapy group for people with learning disabilities and anxiety. Br J Learn Disabil. 2021;49:87–99. https://doi.org/10.1111/bld.12344

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Social Theory and Practice Vol. 49, No. 4 (October 2023): 605–628 DOI: 10.5840/soctheorpract2023817200

Cognitive Disability and Social Inequality

Linda Barclay

Abstract: Individuals with ‘severe’ cognitive disabilities are primarily discussed in philosophy and bioethics to determine their moral status. In this paper it is argued that theories of moral status have limited relevance to the unjust ways in which people with cognitive disabilities are routinely treated in the actual world, which largely concerns their relegation to an inferior social status. I discuss three possible relationships between moral and social status, demonstrating that determinate answers about the moral status of individuals with ‘severe’ cognitive disabilities are neither necessary nor sufficient for defending the imperative that they be treated as our social equals.

Keywords: cognitive disability, moral status, social status

I. Introduction

Individuals with what are usually referred to as ‘profound’ or ‘severe’ cogni- tive disabilities have notoriously been discussed in philosophy and bioethics to determine whether their moral status is higher than that of many nonhu- man animals (McMahan 2002; Singer 2011).1 Disability advocates vehement- ly contest claims that such individuals don’t share the same moral status as other human beings, and moreover argue that suggestions to the contrary are harmful and dangerous (Kittay 2010). Although this debate about the moral status of individuals with cognitive disabilities is very well-rehearsed, nothing approaching a consensus view has emerged. Yet debates about moral status continue to dominate philosophical discussion of people with ‘severe’ or ‘pro- found’ cognitive disabilities, with all sides assuming that the outcomes of such debates are pivotal for determining how we ought to treat such people.

1. From here I will usually just refer to such individuals as ‘people with cognitive disabili- ties.’ It should be understood that debates about the moral status of such people are only relevant to those individuals with the most ‘severe or ‘profound’ disabilities, who consti- tute only a very small proportion of people with cognitive disabilities. There is little con- sensus about the exact nature of the categories ‘severe’ and ‘profound’ cognitive disability, nor exactly which individuals fall into that category. At the very least it is to be understood that I am referring to people with reported very low IQ, who have little to no facility with language (they are unable to speak, read or write) and who need constant daily care with eating, personal hygiene, and avoiding basic threats to safety. For more detail about the broad and diverse group of people often identified as having ‘severe’ or ‘profound’ cogni- tive disability see Vorhaus 2017, Carlson 2019.

Linda Barclay606

In this paper I will argue that theories of moral status have quite limited relevance to the unjust ways in which people with cognitive disabilities are routinely treated in the actual world. What I will show is that this routine un- just treatment threatens the social status of people with cognitive disabilities. After explaining the difference between moral status and social status, I will argue that fixing on moral status plays only a limited role in helping us to un- derstand and respond to the injustice of pernicious social status hierarchies. As such, if we are concerned to address the injustices perpetrated against people with cognitive disabilities, we need to focus much more on neglected issues of social inequality.

My arguments in this paper are a clarion call for us to focus discussion of cognitive disability on issues of injustice. Other philosophers have persuasive- ly argued that we can neither understand nor address racial and sexual injustice by focussing solely on the bad attitudes of individual actors, whether explicit or implicit (Anderson 2010; Haslanger 2015). The sources of injustice and the causal mechanisms that sustain it are far more complex than an individualis- tic focus on the beliefs or other attitudes of agents would suggest. This paper furthers this structural focus by applying some of its insights to the sphere of disability: the sources of injustice and the causal mechanisms that sustain it are far more complex than the almost exclusive focus on beliefs about moral status would lead us to suppose.

In sections II and III I will explain what is meant by moral status and so- cial status respectively. In discussing the nature of moral status in section II, I will explain that the possession of moral status is necessary in order to be harmed by pernicious social hierarchy. As such, establishing that people with ‘profound’ cognitive disabilities have moral status is of first importance to any discussion of the injustice of social status hierarchies. If an individual has no moral status, they cannot be a victim of social injustice either. Beyond that important role, debates about moral status have very little further to add. In section IV I will reject the conventional view which holds that when people are treated as social inferiors it is because they are assumed to have lower moral status. I will argue that there can be a number of reasons for why social hier- archies emerge that have nothing to do with individuals’ beliefs about moral status, and this can certainly be the case with disability.

In section V I will argue that even if social status hierarchies are morally impermissible because they violate respect for people’s moral status, it is still the case that theories of moral status are very uninformative in terms of ex- plaining the nature, function and ultimate impermissibility of social hierarchy. In this sense, fixing on the moral status of people with ‘profound’ cognitive disabilities does little explanatory work in determining how they ought to be treated.

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In section VI I will suggest something that is perhaps more confronting: that we do not need to demonstrate that people with ‘profound’ cognitive dis- abilities possess equal moral status with other human beings in order to de- nounce the unjust ways they are treated in the actual world. The overwhelming majority of us will agree that much of treatment I discuss in this paper is wrong and must be rectified. Yet we can agree on that quite independently of whether we also agree that people with cognitive impairments share exactly the same moral status with more typical adult human beings. To a large extent, that par- ticular issue is entirely orthogonal.

In sum, if we want to understand how people with cognitive disabilities are treated unjustly, understand why they are so treated, and what we must do better, then we must pay far more attention to the nature and function of perni- cious social hierarchies.

II. Moral Status and Its Necessity

Our widespread practices and behaviour suggest that most of us believe that all human beings have a higher moral status than (almost all) animals. Philo- sophical justification for this belief has proven elusive. It is helpful to begin by distinguishing two broad approaches to the question of moral status (cf. Jaworska and Tannenbaum 2014).

Threshold accounts. Questions about the moral status of individuals with ‘severe’ or ‘profound’ cognitive disabilities are unavoidably vexing for any view that designates high level, typically human cognitive wherewithal—ra- tionality, the capacity for moral agency—as a necessary condition for moral status. Moral status is a threshold concept for such views: individuals who fall below some minimal threshold have no moral status at all, and individuals who are at or above the minimal threshold enjoy equal moral status. Human and non-human individuals who lack rationality, the capacity for moral agency, etc., have no moral status at all. We are permitted to treat them as we will, as long as we do not violate any moral requirements towards those who do have moral status (including oneself).

Interest accounts. On these accounts, the concept of moral status, if it is relevant at all (Sachs 2011), refers to identifying the specific individual inter- ests which must be considered in determining how we ought to treat others. In developing a version of this approach, Peter Singer notoriously compares human beings with very ‘severe’ or ‘profound’ cognitive disabilities to non- human animals (Singer 2011: 135–36). Non-human animals and severely cog- nitively disabled humans share many of the same interests, such as that of avoiding pain. Whether individual humans with severe cognitive disabilities have an interest in continued living has also been extensively discussed under

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this approach. Following Michael Tooley (Tooley 1972), Singer argues that only persons can claim such an interest, and most non-human animals, human infants, and some individuals with severe cognitive disabilities are not persons (Singer 2011: 160–67). The wrongness of (painlessly) killing such individu- als, according to Singer, is to be entirely explained by its negative effects on overall utility, that is, on the interests of others, such as parents, would-be parents and so on. Hence interest accounts, like threshold accounts, assuage our shocked sensibilities by suggesting that we have indirect reasons for not wantonly killing (for fun, say) individuals who are not persons.

Despite the obvious differences between threshold and interest accounts of moral status, it is what they share in common that is of interest here: neither draws the boundaries of moral status to include all and only human beings. On the interest approach, there is no compelling reason to deny that most animals will share some of our most important interests, such as avoiding pain and suffering. Equally, not all humans will have the same interests: some humans who are persons will have more and perhaps weightier interests than those humans who are not persons. Most threshold accounts of moral status propose criteria for moral status that will exclude most animals, but also many human beings. In response to the concern of exclusion, one can propose a less de- manding threshold: the capacity for sentience for example, rather than capaci- ties for rationality or moral agency. A criterion like sentience would helpfully include virtually all human beings, but would include nearly all animals as well. Thus, neither approach to moral status defends our common intuitions about the equal moral status of all human beings, and lesser moral status (if any) for most animals. While those who advocate for the interests or rights of animals might embrace this conclusion, it is considered a wholly unacceptable outcome by those who advocate for people with disability: ‘reducing’ people with cognitive disabilities to the status of animals exemplifies everything we rail against.

More recently new theories about the criteria for moral status have been developed with the aim of defending the common belief that all human be- ings share a higher moral status than (most) non-human animals. The various theories are well-rehearsed, such as genetic species membership (Liao 2012), or the capacity to participate in person-rearing relationships (Jaworska and Tannenbaum 2014). All such proposals remain deeply controversial. As Ag- nieszka Jaworska and Julie Tannenbaum argue, “providing an adequate theory to account for the FMS [full moral status] of unimpaired infants and cogni- tively impaired human beings (whether infants or adults) without attributing the same status to most animals has proven very difficult . . . this challenge has not been fully met by any of the existing accounts of the grounds of moral status” (2018).

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This challenge is particularly acute for theories of moral status that focus on the capacities or properties of individuals as grounds for moral status. I will not focus further on the details of such approaches to moral status, nor on whether they succeed in meeting the challenge described by Jaworska and Tannenbaum. Of somewhat more relevance to my argument are alternative approaches to moral status which shift the focus away from the capacities of individuals and on to more social features characteristic of the relations be- tween human beings. There are a range of such views, including those identi- fied as Wittgensteinian in nature (cf. Diamond 1978); those which argue that moral status can be bestowed on human beings (but not animals) by virtue of being taken into the life of the human community (Vehmas and Curtis 2017); and those which argue that what we owe other human beings is related to their participation in characteristically human forms of social life (Vorhaus 2017). As I will argue in Section V, some of these theories do indeed identify some- thing morally crucial, but mistakenly attribute it to the issue of moral status. Understanding the moral importance of participating in specifically human forms of social life does not lead us to conclusions about the bestowment of moral status, but about the need to identify and oppose social hierarchy. This latter conclusion can be reached whilst remaining largely silent on the vexed question about the precise nature of the moral status of people with ‘profound’ cognitive disabilities.

Before proceeding with this discussion, I acknowledge that it is certainly true that the arguments to follow presuppose that people with cognitive dis- abilities have some moral status. Put somewhat differently, the possession of moral status is necessary in order for a person to be wronged by social hierar- chy: if a person has no moral status, nor can they be wronged by social status hierarchies either.

As we have seen, this is a vexing result for many threshold accounts of moral status centred on individual capacities, as many people with very ‘severe’ or ‘profound’ cognitive disabilities will not meet many nominated thresholds. If an individual does not meet whatever threshold is posited for possessing any moral status at all, then we can never have any direct reasons to avoid treating them as social inferiors. I can hardly take on the task here of showing why demanding threshold views are wrong. I hope my discussion of the obvious harms inflicted on people with cognitive disabilities when they are treated as social inferiors acts as a kind of reductio of demanding threshold views. If not, I concede that my audience is restricted to the vast majority of us who believe that people with ‘severe’ or ‘profound’ cognitive disabilities do indeed possess at least some moral status.

In contrast to threshold views, interest accounts of moral status have little difficulty in accepting that people with even the most ‘severe’ cognitive dis-

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abilities have interests and therefore moral status: on virtually all interest ac- counts any individual who is sentient has some interests. Most of my claims about the harms caused by social hierarchy will be accepted by those who hold interest accounts of moral status, including utilitarians, as I will indicate throughout: indeed, for most such accounts, it is precisely in identifying the harms of various forms of social hierarchy that we establish an individual’s interest against them.

On the other hand, as I have mentioned, it is certainly not clear that interest accounts would recognise that newborns have an interest in continued living. Singer has denied that newborns (disabled or not) have a direct interest in continued living (Singer 2011: 135–36). I acknowledge that it is precisely this position that motivates many people to develop alternative accounts of moral status—so that they can demonstrate that it is directly wrong to kill newborns, including disabled newborns. While I agree that it is, all of the harms that I identify in this paper apply only to those who are living among us as socially- engaged individuals. As such, my arguments about social hierarchy do not obviously apply to the case of newborns. I must confess, I am not overly con- cerned by this limitation. While I appreciate the motivation some have to ad- dress utilitarian views about the moral status of newborns, part of my motiva- tion in this paper is to address what I take to be the comparative neglect of the way we engage with the millions of people with cognitive disability who live among us, and always will. For example, we don’t talk nearly enough about the dirty business of residential facilities in moral philosophy.

III. Social Status

According to distributive theories of justice, equality consists of people hav- ing an equal amount of something, such as resources, capabilities or opportu- nity for welfare (Arneson 1989; Cohen 1989; Dworkin 2000). Numerous and elaborate theories of distributive justice have been developed as answers to the ‘equality of what’ challenge since the 1980s.

Social egalitarianism has emerged partly in response to the dominance of distributive approaches to equality. Social egalitarians deny that equality ex- clusively concerns what one gets in the form of individual holdings. Equality also, or perhaps more fundamentally, concerns the quality of the relationships between individuals, including between individuals and those who govern them. It has been argued not only that distributive approaches have ignored these relational considerations, but that numerous distributive theories propose conceptions of equality that promote the kinds of relations that social egalitar- ians oppose. For example, both Elizabeth Anderson and Jonathan Wolff argue that the focus on individual responsibility in many distributive theories would

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endorse disrespectful and disparaging judgments which promote social hierar- chies (Wolff 1998; Anderson 1999).

What exactly are the kinds of relations that exemplify social equality ac- cording to social egalitarians? To a large extent, social egalitarians develop their case by way of negative example, where we are encouraged to share their intuitions that a particular form of relationship is at odds with social equality. Moreover, social egalitarians have been slow to develop their own systematic accounts of equality with the detail and complexity characteristic of distribu- tive approaches.2 As such, we’ll begin with some of the paradigmatic examples that social egalitarians offer as exemplifying problematic instances of social hierarchy. Examples of hierarchical social status relations typical in class and caste societies are commonly cited. Social inferiors might be expected to en- gage in explicit acts of deference and submission, such as tugging at the fore- lock, bowing and avoiding eye contact, whilst superiors might condescend, sneer and belittle and expect to receive various social privileges. Members of highly stratified societies also tend to lack easy, fraternal relations when their tastes, leisure pursuits, consumption patterns, assumptions, stereotypes and sources of information are strongly divided according to location in the social hierarchy. The vision expressed by recent social egalitarians “identifies a so- cial ideal, the ideal of a society in which people regard and treat one another as equals, in other words a society that is not marked by status divisions such that one can place different people in hierarchically ranked categories, in different classes for instance” (Miller 1997: 224).

Social status hierarchies are also connected to domination. Some exercise considerable control over others, including decision-making authority at work and in the provision of public services, decisions over what is produced and consumed, what achievements are celebrated and how prizes are distributed. Social inferiors are often marginalised from society in various ways, either locked out of highly rewarded and esteemed productive activity, or isolated from valuable forms of social and cultural life. Individuals in highly stratified societies also experience radically different access and success with respect to political power (Young 1990). The effects of domination, marginalisation and unequal power can range from feelings of worthlessness and shame, lack of access to opportunities for esteem and personal autonomy, lack of opportunity for economic, social, political and cultural engagement and influence, to sig- nificantly poorer health outcomes (O’Neill 2010).

We can render some of this somewhat abstract discussion more concrete with an example. My focus is on the case of people with cognitive disabilities, but examples abound of groups stratified by gender, class, race or other mark-

2. In section V I will argue that these are not flaws of social egalitarianism, but indicate a pur- poseful methodological approach with superior capacity to address real world inequality.

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ers of social hierarchy. People with cognitive disabilities often share many core experiences of social status inequality. Imagine one such group of people with a range of disabilities living in a supported residential environment. Some of the care workers speak to them with a tone of condescension and infantiliza- tion whereas others routinely express impatience and contempt. The privacy of the residents is often violated, when staff walk into their rooms without knock- ing, or assist them with personal care without ensuring others cannot interrupt or observe. The food and recreational activities offered to the residents are of poor quality, and inadequate staffing levels also contributes to residents often missing out on showers, assistance with eating and participation is social and cultural activities. To keep costs down, the facility is located on the outskirts of town and difficult to access via public transport. To meet their safety reporting targets, residents are not permitted to leave the facility on their own or even to wander the gardens unattended. Many of the residents therefore have little to no contact with close friends or family members, nor are they able to engage in the life of their community. Many of the residents are terribly lonely, bored and frustrated, are sometimes hungry, and suffer occasional medical problems associated with a lack of hygiene and prompt medical attention. No one takes initiative to ensure that residents have a genuine opportunity to vote in elec- tions. While much of this story will resonate with those familiar with residen- tial care for people with cognitive disabilities, it will also be familiar to many older people living in supported accommodation.

Does the treatment the residents are exposed to—the behaviours, the prac- tices, the policies—ensure that they enjoy equal social status with those they routinely engage with? I don’t think so. Those who are charged with support- ing them often act as social superiors, free to express scorn, and to infantilise. Given the vulnerability of the residents with respect to accessing the most ba- sic of requirements such as food, personal care and social contact, it is unsur- prising that some of them who are less disabled might tend towards deference and servility and a general desire to please. The residents are marginalised, or indeed segregated, from the broader life of the community, its cultural, so- cial and political activities. They can be aptly described as dominated when they have no security in staying clean, or consuming nutritious meals, but are instead at the mercy of others’ decisions around staffing levels, staff qualifica- tions and institutional priorities. The residents and those who have such ex- tensive control over their lives do not “stand in front of each other as equals” (Bidadanure 2016: 236).

The kind of social status hierarchy I have described is very widespread in residential care settings (Gjermestad, Luteberget et al. 2017; Steele, Swaffer et al. 2019; Murphy and Bantry-White 2021). As Kieran Murphy and Elea-

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nor Bantry-White state, their evaluation of over 620 inspection reports of care homes in Ireland demonstrates

that people with an intellectual disability were not regarded as citizens capable of inclusion in society. If they were, people would not have had to experience daily restrictions on their lives, be subjected to abuse and be segregated, isolated and neglected. . . . The findings depicted a system of residential care that disempowered, controlled and monitored people. The combined findings demonstrated that the system of residential care for people with an intellectual disability was one of total control as described by Altermark in his work Citizenship Inclusion and Intellectual Disability, namely a system designed to monitor behaviour; dependant on a hierarchy between staff and residents; constant control; with the control being individualised to micromanage every aspect of people’s lives. (Murphy and Bantry-White 2021: 763–64)

Moreover, it is plausible that such extensive status differences render residents vulnerable to further forms of maltreatment and abuse. We know that vulner- able people in residential care suffer high levels of sexual and physical abuse (Baladerian 1991; Horner-Johnson and Drum 2006; Murphy and Bantry-White 2021). Condescension, neglect, powerlessness and infantilization, contribute to an environment where people relegated to an inferior position in a social hierarchy are prey to violence and egregious forms of harm. I will elaborate on this claim in more detail in the next section.

It emerges from this picture that one’s location in a social hierarchy is constituted by the nature of the relationships one has with others. One occupies a lower position in the social hierarchy because of the way one is regularly treated by others, and the decision-making power they yield. The ideas of reg- ularity and scope are important here. Everyone can be occasionally subject to sneering and condescension by particular others. This is not sufficient to make one an inferior in a social status hierarchy. Such inferiors are regularly subject to a range of convention and norm-driven behaviours of this type. Similarly, each of us can occasionally find ourselves marginalised or excluded from a valuable opportunity through happenstance, bad luck, our own earlier deci- sions, etc. Social inferiors are marginalised and excluded from a wide range of economic, social, political and cultural opportunities which has a very signifi- cant impact on the course of their lives.3

Having now outlined the basic contours of both moral status and social status, questions arise as to the nature of the relationship between them. Given that most philosophical debate about people with ‘profound’ cognitive dis- abilities concerns moral status, it would be natural to assume that questions 3. It follows that there can be vagueness and disagreement in some cases as to where people

are located in social hierarchies, or whether there are any hierarchies at play. I think this is just what we should expect. In marginal cases, there can be genuine disagreement as to whether social hierarchies exist and as to where people are located. I discuss some of this complexity in (Barclay 2020).

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about the nature and permissibility of social status hierarchies are subordinate to the question of moral status. In one respect, this is true: as I have explained in Section II, people with ‘profound’ cognitive disabilities must possess some moral status in order for these behaviours to be directly morally troubling. The possession of moral status is a necessary condition for the impermissibility of social hierarchy. Those who subscribe to demanding threshold views of moral status will find no direct reasons to morally condemn the harms and suffering inflicted on the residents. As I have stated, those who hold such views are not my audience here. On the other hand, most interest accounts of moral status will surely believe that significant interests are at stake in these examples. People with cognitive disabilities can be lonely, bored, frustrated, agitated, humiliated, hungry, and vulnerable to the harms of abuse and medical neglect. As such, some of their significant interests are at stake in pernicious social hierarchies. Utilitarians like Singer certainly agree that individuals with cog- nitive impairments can suffer in the ways I have described, and as such they have morally-considerably interests that must be taken into account. They have moral status.

Beyond recognising that people with ‘profound’ cognitive disabilities do possess moral status, theories of moral status have little further to add to ques- tions about the nature, function and permissibility of social hierarchies, as I will argue in the next three sections.

IV. The Conventional View

The most straightforward way to suggest a more significant role for theories of moral status in addressing the morality of social hierarchy is by adoption of what I will call the conventional view. According to the conventional view, it is the belief that some people have lower moral status that causes social hier- archy: in other words, the various attitudes and behaviours that constitute so- cial status hierarchies express people’s beliefs that social inferiors have lower moral status.

The conventional view is perhaps the simplest way to understand some of the following claims by prominent social egalitarians. Anderson states that “Inegalitarianism asserted the justice or necessity of basing social order on a hierarchy of human beings, ranked according to intrinsic worth. . . . Egalitar- ian political movements oppose such hierarchies. They assert the equal moral worth of persons” (Anderson 1999: 312). Similarly, Christian Schemmel sug- gests that “[egalitarian] movements generally demand treatment that affirms their equal moral status. What they are after is the confirmation that the people they represent are not, by virtue of belonging to a group such as women or gays, of inferior moral worth, and, accordingly, they demand state action that

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makes this clear” (Schemmel 2011: 134). I suspect the conventional view un- derpins much of the objection to social status hierarchies, taken as they are as expressions of the belief that some people have lower moral status or worth. Indeed, so widespread is this view that most people do not typically distinguish between moral and social status when denouncing inequality and hierarchy.

I certainly have no wish to deny that some of the most egregious social status hierarchies have been built upon and sustained by widely shared beliefs that those at the bottom of such hierarchies have lower moral status. This is certainly the case for disability: people with disabilities have been treated in- tolerably because of widespread dehumanising stigma around disability. Noth- ing in what follows should be taken to deny this fact. Nevertheless, there are numerous factors that sustain hierarchical social relations which cannot be re- duced to the existence of widespread beliefs about inferior moral status, and this is also the case for disability. I will argue that beliefs about low moral sta- tus are not necessary for social hierarchies to emerge; indeed, social hierarchy can emerge even when people sincerely disavow the view that some people are of lesser moral worth.

To begin with the relatively mundane, social hierarchy can sometimes emerge for reasons that are themselves morally acceptable. Decisions about the allocation of limited resources, planning decisions, health care priorities and the like can all have the unintended and often unforeseen effect of con- tributing to social status hierarchies. Consider a concrete example of children with cognitive disabilities who are taught separately from children without such disabilities. Segregated schooling might be a response to concerns from parents and educators about the failure of children with disabilities to thrive in mainstream schools. There may be some evidence to support this claim, and concern from parents about the bullying and social exclusion their children have been subjected to. Evidence might suggest that educating some children with cognitive disabilities in specialist schools improves their educational and social outcomes. Nonetheless, the evidence might also show that segregated education is a contributing factor to longer-term marginalisation, as people without disabilities remain ignorant of the capabilities of people with disabili- ties, as well as insecure and uncomfortable in interacting with them.

Consider also the case of residential aged or disability care located on the outskirts of major cities. Planners and policies makers have to allocate scarce resources, and residential homes in the inner city might be prohibitively ex- pensive. Planners may also be rightly concerned that the high cost might put inner-city facilities out of reach of all but the wealthy. As explained above, locating residential facilitates far from city centres or from public transport can lead to marginalisation, but in this case being marginalised would not neces- sarily be caused by the belief that people in need of residential care have lower

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moral status. These examples—and there are many just like them—support the contention that social status hierarchy can sometimes arise from a combina- tion of factors other than a widespread belief that some people have low moral status.

More generally, individuals’ beliefs often do not provide the best explana- tion for the persistence of social status hierarchies. Sally Haslanger has argued that ‘social meanings’ can create social injustice, where those meanings con- sist of more than beliefs (Haslanger 2015; Haslanger 2017a). She argues that the way we think and act is shaped by culturally-shared meanings—“cultural schemas”—beyond and sometimes prior to propositional attitudes. They can include things like culturally shared propositions, but also norms and concepts that when internalised by individuals become the basis of various behavioural and emotional dispositions. They highlight some parts of the world and ob- scure others, thus partly shaping our perception; they also link some things to others, conceptually, or through shared narratives and stories. “Thought, perception, emotion and other psychological states depend on a public ‘field of preexisting meanings’ . . . this ‘field’ shapes and conditions our experience and agency, and provides a kind of palette of psychological content” (Haslanger 2017a: 154). Such cultural schemes function to provide us with the ‘tools’ for interpretation, interaction and coordination that enables social fluency with respect to our shared practices. Cultural schemas shape the way we respond to the world, how we organise ourselves, distribute resources, use implements and objects, relate to one another, formulate rules and policies. The effect is that our social arrangements and practices reinforce the social meanings that produced them, making it seem as though our cultural schemas interpret and value the world the way it really is. Culture is a source of beliefs, not just an effect of them (Haslanger 2017b).

One upshot of Haslanger’s view is that we underestimate the resilience of social injustice if we characterise it simply as an effect of pernicious or false beliefs. Injustice can be sustained by practices, and the social structures they constitute. Returning to the example of residential care, care staff im- mersed in shared practices of caring for the disabled and the very old often take themselves to be acting in the best interests of those they care for, fully dedicated to securing their well-being, avoiding risk, improving accessibility and showing kindness. The social meanings at play are internalised by carers to shape their shared understandings of what constitutes good caring, allowing a degree of fluency in their coordination. Challenging some of their beliefs (for example, about risk, privacy, dependency, appropriate tone of voice) is likely to be of limited benefit when the practices they engage in reinforce the inter- nalised social meanings that inform them. To overemphasise the role of perni- cious beliefs in producing or sustaining social hierarchy is to ignore the more

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complex structural story which can explain the way an unjust social system reinforces itself. It can do so even in cases where people disavow pernicious beliefs about the moral inferiority of others. As Haslanger argues, changing patterns of thought, both explicit and implicit, often requires changes to social structures and the schemas that inform them (Haslanger 2015).

Indeed, insofar as people may hold beliefs about the moral inferiority of disabled people using residential care, such beliefs might be an effect of wide- spread practices which perpetuate social status hierarchies, rather than their cause. It is plausible to suppose that the creation of social hierarchies between people with cognitive disabilities and those who interact with them is some- thing that can contribute to shaping even more unjust practices. We cannot ignore not only the long history, but also contemporary evidence of the sheer extent of injustice to which people with cognitive disabilities are exposed, including violence, extreme physical, medical and emotional neglect, sexual abuse, institutionalisation in squalid and unsafe environments and involuntary medical experimentation, are just some examples (Baladerian 1991; Horner- Johnson and Drum 2006; Troller, Srasuebkul et al. 2017). The construction of people with cognitive disabilities as social inferiors surely plays some explan- atory role in such maltreatment (cf. Carlson 2019). To routinely engage with people with cognitive disabilities as social inferiors can contribute to dehu- manising social meanings around disability that in turn shape practices that all of us should recognize as profoundly unjust—those characterised by violence, sexual abuse, severe neglect, and distributive injustice.

I have argued in this section that beliefs about lower moral status do not necessarily explain the emergence or persistence of social inequality. Social hierarchies can emerge from and be sustained by a range of beliefs and broader social meanings, some of which may not themselves be morally troubling, or at least not obviously so. The emergence and persistence of social status inequal- ity is often not aptly described as an expression of widespread beliefs about inferior moral status, although the existence of widespread social status hier- archies can itself play a causal role in producing and perpetuating views about the inferior moral status of people with cognitive disability. It is of course true that some of the most egregious social hierarchies have been undergirded by widespread beliefs about the inferior moral status of some groups of human beings. Nothing argued here should be taken to deny that fact. What is being argued is that the conventional view is too limited to fully explain the extent of the social hierarchy characteristic of the relationship between people with and without disabilities.

One implicit upshot of the argument so far is that we need to be far more attentive to the social facts before any conclusions can be drawn about the role of moral status claims in perpetuating social hierarchies. Only by paying

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attention to the actual history, cause, nature and justification of various social relations can we make judgements about what role, if any, beliefs about moral status are playing. This appeal to the importance of social facts is central to the methodology of much recent social egalitarian theorising, which I will elabo- rate upon in the next section.

V. The Impermissibility View

I have rejected the view that social status inequality is always just an effect or expression of the belief that those at the bottom of the hierarchy have lower moral status. Another possible relationship between moral status and social status is what I will call the impermissibility view. According to the imper- missibility view, social status hierarchies are morally impermissible because they fail to respect our moral status. When Anderson claims that egalitarian movements “assert the equal moral worth of persons” and Schemmel says that they “demand treatment that affirms their equal moral status” they might be interpreted as stating that social status hierarchies are morally wrong be- cause incompatible with our equal moral status. This normative claim does not presuppose the conventional view, which I criticised in the previous section. One can accept that social status hierarchy might arise from factors other than beliefs that some have inferior moral status, but nevertheless argue that social hierarchy must be eliminated because it is in conflict with our moral status. In other words, the fact that all human beings have moral status is sufficient to denounce social status hierarchy, whatever its causal origins, and whatever the forces that sustain it.

I am largely sympathetic to this claim. I think many cases of social hier- archy are morally impermissible because they do not respect our moral status, even if they happen to have arisen from reasons or circumstances that are in themselves morally acceptable. However, I shall develop two arguments in what follows. Firstly, in this section I will show that facts about moral status do very little explanatory work in establishing this case against social status hierarchies. In terms of identifying morally unacceptable social hierarchies, and knowing how to rectify them, philosophical views about moral status are largely uninformative. To understand when social hierarchies are wrong be- cause in conflict with moral status, we need to pay much more attention to the nature and function of social inequality. In this sense, further theorising about the grounds for moral status is rather irrelevant. Secondly, in section VI I will also argue that it is not necessary to establish that all participants in social relations must share equal or the same moral status in order to plausibly denounce social status hierarchies. Even if some people have different or even ‘lower’ moral status than others, social status inequality can still be morally

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impermissible. As such, we can reject much of the injustice inflicted on people with ‘profound’ cognitive disabilities in the actual world without establishing that we all share the same moral status.

What would be required to determine whether social hierarchies are mor- ally impermissible? Certainly not all social hierarchies strike us as impermis- sible. Social relations are replete with behaviour that expresses social hierar- chy. When a judge enters a courtroom, we stand for her; we address her only by title, never by name; we respect her almost unlimited authority in the court to dictate when we may or may not speak, stand, and so on. Another example: in most countries, there are numerous ways in which children and the young are expected to behave deferentially toward (some) adults. They too might be required to use titles when addressing others, or to ask permission to speak or leave a room. Perhaps they have to stand when a teacher enters the room. Most of us consider it acceptable to talk about babies to others even when the baby is in the room, to ignore adult standards of privacy when bathing the baby, and to use a tone of voice that would be regarded as shockingly condescending if used in conversation with an adult.

Are these instances of morally unacceptable social hierarchy? We will not get far in answering this question by arguing that non-judges, young people and babies have full moral status, for example because they are members of the human species, or are sentient, or can participate in person-rearing rela- tionships, or because they are rational (admittedly a stretch in the case of ba- bies!). So what? What we need to know is whether non-judges, young people and babies have a significant interest in not being treated in the manner de- scribed, and it strikes me as fairly obvious that the answer to this question must be determined by careful attention to the social facts. Most obviously, we would need to be attentive to various facts about whether these modes of treatment cause harm to the recipients; whether broader social damage is wrought by allowing relations marked by such status differences to flourish; or whether these particular behaviours and norms interact with other aspects of social relations to generate harmful and damaging outcomes. For example, we might ask whether our norms in the courtroom hinder fair trials; whether such norms contribute to judges wielding disproportionate political power, or whether they create or reinforce expectations that others should defer and grovel to judges in a wide range of social settings outside of the court room. We might ask whether such norms induce feelings of shame and humiliation in other participants in court cases, and whether they contribute to widespread stigma against lawyers, jurors and defendants who are required to stand for the judge and refer to her as ‘Your Honour.” Even if the answers to some of these questions are affirmative (which I very much doubt), the case against our court room norms of status hierarchy is not established. For we would still have to

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ask whether there are compelling reasons for the norms, and whether it is pos- sible to abolish or modify them without jeopardising respect for those reasons.

Similar questions arise in scrutinising the behaviour between adults and children. Does bathing babies naked in front of other people undermine mor- ally valuable relations between babies and adults? Is it likely to induce shame and humiliation, or contribute to a broader pattern of social meaning and be- haviour which render babies vulnerable to physical and psychological neglect and harm? Posing questions such as these takes us deep into an exploration of the nature of babies, how they flourish, and facts about how our social norms and patterns of behaviour might stunt or facilitate such flourishing. It requires attention to the construction of social meaning and how cultural schemas can sustain our participation in social injustice.

Theories of moral status are almost entirely irrelevant to this task. Take the threshold account of moral status. Let’s grant for the sake of argument that all non-judges and all young people and babies meet the threshold for moral status. That starting place affords no guidance as to whether the behaviours we are discussing are morally acceptable or not. Jaworska and Tannenbaum, address head on the question of what follows from any given threshold ac- count of moral status in terms of how we should treat one another. They come up with the following suggestions, all of which they concede are contentious, some highly so: a moral presumption against certain types of interference, such as killing and medically experimenting on the being in question; a strong, but not necessarily stringent reason to aid; a strong reason to treat fairly, with the caveat that what counts as ‘fairly’ is highly contentious (Jaworska and Tan- nenbaum 2018). None of these questions figure directly in a serious explora- tion as to which of our social status examples are impermissible.

An interest approach to moral status will have a much more direct path to acknowledging that all nonjudges have an interest in avoiding stigma, humili- ation, and harm. We still do not know whether the norms of the courtroom are morally bad or suspect: only close attention to the social facts can guide us. To repeat, we’d have to explore whether the hierarchical norms of the courtroom contribute to harm outside of it, by, for example, contributing to social mean- ing about the comparative worthlessness of nonjudges which ultimately harms their interests. With babies, the questions are more complex still: whether they even have an interest in avoiding powerlessness and marginalisation, what their actual interests might be, and what patterns of behaviour are consistent with promoting them. To reach any conclusions on these vexed questions re- quires that we pay attention to how social hierarchies actually function in real- world cases. Knowledge about how social hierarchies function is essential to reaching any normative conclusions about when we need to avoid them, which ones specifically we need to avoid, and how we might do so. In this sense, un-

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derstanding that we have the moral status granted to us on an interest account is not sufficient to establish the moral impermissibility of social hierarchy, or even throw much light on why it is so.

Critics of social egalitarianism have accused it of being vague and impre- cise in its approach to equality. The accusation is based in a number of reasons, including the tendency of social egalitarians to eschew ideal theories of justice of the type that have dominated distributive approaches to equality. Instead of engaging in endlessly complex specifications of an ideal conception of equal- ity, social egalitarians often proceed by highlighting actual cases of manifest injustice: indeed, they are often very sceptical that ideal theories of justice shed much explanatory light on, or prescriptive guidance with respect to, ac- tual cases of injustice (Anderson 2010; Anderson 2012; Wolff 2015). Their preferred methodological approach is to scrutinise the nature and function of actual instances of social inequality as an essential step in proposing effective solutions. As such, social egalitarians have typically been highly attentive to the actual ‘sociology of disadvantage’ (Axelsen and Bidadanure 2019). As Da- vid Axelsen and Juliana Bidadanure argue, the diagnostic precision attained by social egalitarians (their ability to identify ills within the actual world) “stems from constructing their account around, and not abstracting from, the most sa- lient features of how inequalities and disadvantages actually appear and func- tion” (2019: 344, emphasis in the original). Or as they put it, the normative significance of equality is tied to the sociology of actual disadvantages.

If we return to the cases which are the focus of this paper, we need to ask a range of questions about how inequalities function. Does speaking with condescension to adults with cognitive disabilities, or ignoring privacy norms, humiliate them? Even if some such people are impervious to feelings of hu- miliation and shame it is equally crucial to know whether such behaviours contribute to pernicious social meanings around disability that can render dis- abled people vulnerable to neglect and harm. Does housing people with cogni- tive disabilities away from the centre of communities lead to social isolation, loneliness and vulnerability to abuse? Do paternalistic policies in residential settings rob people of the opportunity to develop independence skills, and thus decrease their opportunities for self-esteem and the satisfaction of their preferences? Does the inability to contribute to the life of the community— through work, social, cultural and political engagement—render them prey to the preferences and choices of others in which their own needs are likely to be overlooked? I am confident that the answers to these questions are often af- firmative, and that as such any interest approach to moral status will grant that important interests are at stake. But it is not my intention to prosecute this case here. What, instead, I have been arguing is that if philosophy is to contribute to detecting and addressing the actual injustices to which people with cognitive

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disabilities are most often exposed, we need to spend much more time thinking about the nature and function of social status rather than engaging in endless abstract debates about the grounds for moral status.

As we know, many philosophers who focus on theories of moral status trade heavily in comparing people with ‘profound’ cognitive disabilities to animals. As such, they are likely to do so here as well by asking: if it is of- ten wrong to treat people with cognitive disabilities according to hierarchical social norms, doesn’t it follow that it must also be wrong to treat nonhuman animals in similar ways? One of the most significant advantages of paying at- tention to the actual facts of social inequality is that we can cut off any easy comparison between animals and people at the pass. If we want to question the moral acceptability of some of the standard ways we fail to treat animals as our social equals—commanding them to sit when they’d rather stand; failing to dress them; making them eat from a bowl on the floor—only attention to the social facts will help us. My own view is that the sociology of disadvantage and injustice is likely to reassure us that treating animals in these ways is com- patible with respecting their interests: shame and humiliation are not likely at stake, and nor are the social meanings reenforced by such behaviour likely to lead to domestic pets being abused or neglected. But my aim here is not to prosecute the details of this particular case. What I am pressing is that attention to the nature and function of social relations is what will largely determine the matter, rather than appeal to a theory of moral status. I am assuming that my readers share some of my moral qualms about the cognitive disability cases. In these cases it is clear that theories of moral status have marginal informational importance in determining whether or not relations characterised by social hi- erarchy are morally unacceptable, and nor do we need to rely on the develop- ment of such a theory to tell us why we often have compelling reasons to treat other human beings differently to how we treat domestic pets.

I flagged in section II that there is a degree of overlap between my argu- ment in this paper and more recent theories of moral status that shift focus away from the capacities of individuals toward the social relations between human beings. For example Simo Vehmas and Benjamin Curtis argue that moral status is bestowed on individuals with serious cognitive impairments by virtue of their relationship to other human beings, specifically by being taking in to the human community. Being taken into the human community means being treated as a human within the human community (Vehmas and Curtis 2017: 510), something usually not possible or desirable with animals. Clearly I agree with them that human beings quite appropriately relate to each other differently to how we relate to other animals. But while this aspect of their view is compelling, what they fail to do is offer a convincing reason as to why being taken into the human community in this way thereby bestows moral

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status on human individuals who would otherwise lack such status.4 On my view, the connection between moral status and human relationships is quite straightforward: as long as an individual can suffer harm or experience goods, then we have strong reasons to denounce some forms of social relations and promote other forms. For human beings, many goods or interests can only be promoted within specific non-hierarchical types of human relationships. Moral status comes first: because a human being (already) has various interests, we need to assess which modes of human social interaction promote those inter- ests and which jeopardise them. Because animals also have interests, we need to do the same for them, although their interests are likely to be promoted by very different types of relationships with humans. This is the relevance of human relationships—their centrality to promoting or jeopardising interests— not their ability to mysteriously bestow moral status on individuals who would otherwise lack it.

Closer to my view is an argument developed by John Vorhaus (Vorhaus 2017). Vorhaus also connects moral status to participation in practices distinc- tive of specifically human forms of life. He too defends a very rich account of how our capacities—cognitive, social, emotional—are acquired within and developed through participation in specifically human culture, including the more specific norms and practices that distinguish one human culture from another. What we owe one to one another is connected to such participation, although exactly how participation grounds moral claims of this kind Vorhaus does not say. His primary focus is to bolster the case that human beings par- ticipate in characteristically enculturated human forms of life that animals do not and cannot participate in (for the main), and this despite the fact that some animals may have greater cognitive capacities (of some kinds) than some hu- man beings.

Although he declines to develop a fulsome argument for how such partici- pation is connected to moral status, I would argue that the reasons are already embedded in his account, but that, as with Vehmas and Curtis, stirring the idea of ‘moral status’ into the mix is needlessly obfuscating. In discussing the case of human individuals whose capacities for participation in human culture might be questioned, Vorhaus presents two compelling arguments for why we should treat such individuals as though they have the potential to participate, and thus engage them educationally, socially and in other ways characteristic of human life, as far as possible. Most importantly he says that “there is a large risk to people who are excluded or marginalised from the rest of their society that they will become vulnerable to abuse or neglect, or that they will

4. In another article, Curtis and Vehmas deny that we are required to develop a theory of moral status to defend out commonsense view that all human beings share a higher moral status than animals (Curtis and Vehmas 2016).

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be stigmatised, or that they may be denied their lawful rights. The effects of being treated as an outsider, or as sub-human, or as a non-citizen, are often disastrous and lifelong” (Vorhaus 2017: 73). Secondly, he also argues that if we treat people with cognitive disabilities as though they have the potential to participate, then they are more likely to develop the capacities to do so, and giving up on their potential means they will never develop such capacities.

I certainly agree with Vorhaus’ claims here, but what he doesn’t seem to appreciate is that these arguments already explain why we should draw all human beings, as far as possible, into morally worthy modes of social engage- ment, whether their capacities are actual or merely potential: namely, failing to do so will almost certainly jeopardise their most basic interests, in avoiding harm, cruelty, medical neglect, social isolation, squalor and malnutrition, and so on. Once again, moral status comes first: it is precisely because (nearly all) human beings have such basic interests that their involvement in certain social relations characteristic of human social life (and protection from other relations) is so important for them. There is no additional, extra argument re- quired to explain how participation of this kind grounds moral status. Talk of moral status is obfuscating. We do better by thinking simply about what we owe such people, as Vorhaus suggests: what we owe them is protection against great harms and suffering, and it is by engaging in good social relations, and being protected from bad social relations, that we go a long way to securing such protection. Or so I have argued with respect to the ills of pernicious social hierarchies.

VI. Equal Moral Status?

Much of the discussion so far has suggested a range of ways in which the interests of people with cognitive disabilities can be threatened by relations of social hierarchy. When such hierarchies can render them lonely, bored, dis- tressed, agitated, hungry, vulnerable to abuse and medical neglect then pro- ponents of interest accounts will accept my arguments that if social hierarchy causes or contributes to such harms, then social hierarchy is (ceteris paribus) wrong because it violates the interests and thus moral status of the individuals involved. Utilitarians will accept these conclusions too, as long as the interests at stake are not outweighed by the equally weighty interests of others.

However, such an interest-based argument against social hierarchy need not presuppose that all individuals have equal moral status in the sense that they have the same interests. As Singer has argued, equal interests must be weighted equally, but this is compatible with different individuals having very different interests and some having fewer than others.

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Given that the interest account of moral status is not a strict equality view, it is also possible that not every individual has the same interests in always avoiding hierarchical social relations. Indeed, that has been explicit, for ex- ample, in the discussion of babies, where public naked bathing and a high- pitched tone of voice may promote their interests, whereas we may have good reason to suppose that the exact same behaviour may not promote the interests of adults with cognitive disabilities, depending, as I have argued, on the ef- fects and function of such relations. I certainly wouldn’t rule out that in order to respect the interests of some individuals with cognitive disabilities we may have to engage with them in a way that might resemble the way social inferiors are treated. For example, it may be that a person with cognitive disabilities is comforted by a tone of voice that might sound condescending, that such a voice soothes and relieves agitation. A person with advanced dementia might be wholly incapable of choosing what she wears, consenting to social activi- ties and so on, such that carers make all of these decisions without consulting with her. In cases like these, forms of interaction characteristic of that between social equals may not be in the interests of the individuals concerned. As such, whilst possessing moral status is a necessary condition for rejecting social hi- erarchy, it is not sufficient: everything will depend on the social facts.

If this is right, then it indicates another way in which we have exaggerated the importance of theories of moral status: it is not necessary to show that all human beings share the exact same moral status to compellingly denounce the morally unacceptable ways in which people with cognitive disabilities are routinely treated. If we have good reasons to suppose that treating such indi- viduals as social inferiors contributes to their being significantly harmed, then any interest approach to moral status can recognise and morally reject such treatment, including those interest accounts that would argue that people with severe cognitive disabilities do not have all the same interests as more typical adult human beings.

VII. Conclusion

I have suggested that many of the morally troubling ways we treat people with cognitive disabilities in the actual world involves treating them as social inferiors. I have argued that social status is distinct from moral status and that to determine when relations of social hierarchy are morally impermissible we need to scrutinise their function and effects rather than delve even deeper into familiar debates about the moral status of people with ‘profound’ cognitive disabilities. An appreciation of the fact that people with cognitive disabili- ties have moral status—as they do—does not provide much information about what causes and sustains social hierarchy, nor in what ways it damages, harms

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and oppresses people. That an individual has moral status is certainly a neces- sary condition for the direct impermissibility of social hierarchy, but determin- ing whether or not such hierarchy is actually morally impermissible requires attention to the social facts. If philosophers want to contribute to addressing the myriad of unjust ways that people with cognitive disabilities are so often treated in the actual world, we should recognise that scrutiny of social sta- tus will often be more fruitful than further engagement in intractable debates about the best theory of moral status.

Monash University [email protected]

Acknowledgements I would like to thank the referees for this journal as well as the participants at the “Dignity, Equality and Social Status” conference (Monash University, December 2019) for their very helpful feedback on earlier versions of this paper.

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Research Article Cognitive Therapy for Children with Intellectual Disabilities: A New Look at Social Adaptation Skills and Interpersonal Relationships

Gulnaz Kulnazarova , Zhamiliya Namazbaeva, Laura Butabayeva , and Lazzat Tulepova

Department of Special Education, Abai Kazakh National Pedagogical University, Almaty 050010, Kazakhstan

Correspondence should be addressed to Gulnaz Kulnazarova; [email protected]

Received 12 November 2022; Revised 26 January 2023; Accepted 4 March 2023; Published 3 April 2023

Academic Editor: Marta Pérez-de-Heredia-Torres

Copyright © 2023 Gulnaz Kulnazarova et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

The purpose of the study is to consider the factors influencing the development of the culture of interpersonal relationships and the effectiveness of the influence of cognitive therapy on improving social adaptation skills. The method of cognitive therapy included several types of activities. The first type of activities included group sessions, during which the weaknesses of the interaction and their correction were identified. The second type was conducted in the format of teaching standard models of interaction between people. The third type of activity aimed to teach children to build a dialogue with each other and express their desires and emotions. Classes were held 3 times a week throughout the year. The study was attended by pupils of 5-7 grades of Zhanuya boarding school and special correctional boarding school No. 7 for children with intellectual disabilities. The results of a randomized study demonstrated an increase in the indicators of teamwork skills, self-control, emotional stability, and independent decision-making. The indicators of cheerfulness, openness, sociability, and logical thinking also improved. But the factor of antisocial behavior decreased. All of these indicators directly affect socialization. This strategy can be applied in practice in various specialized boarding schools and schools for children with intellectual disabilities.

1. Introduction

The study of the behavior of children with intellectual dis- abilities is a rather relevant topic today. A decrease in intel- lectual abilities can occur due to various diseases and factors. The basis of pathogenesis is central nervous system disorders [1]. These can be chromosomal pathologies such as Down, Patau, and Edward syndromes. Also, chromosomal abnor- malities include Angelman syndrome and Prader-Willi syn- drome. The features of chromosomal abnormalities include not only delayed intellectual development but also somatic abnormalities. These include sinistral immunity, visual problems, and abnormalities of the nervous system [2]. Abnormalities in mental development can also occur due to complications during pregnancy and childbirth, as well as due to external factors, for example, if the mother had TORCH infections during pregnancy or was exposed to high ionizing radiation doses. Quite often, birth trauma and fetal

asphyxia can be the causes of developmental delay. A nega- tive factor can be the mother’s use of alcohol and drugs dur- ing pregnancy [3]. Also, a decrease in the level of intelligence is observed in the absence of proper education and training of children in their first years of life, this often happens in dysfunctional families [4]. These children often have com- munication problems with the people around them. First, due to a decrease in intelligence, children are not always able to formulate and express their thoughts clearly, which often leads to misunderstandings [5]. Secondly, emotional adapta- tion is impaired, which is characterized by the inability of children to adequately respond to various situations and can also be manifested through isolation and avoidance of social contacts [6]. Due to these factors, most children with intellectual disabilities have problems with socialization and self-realization in society. To help these children, pro- grams are often used to develop their social and communica- tion skills. They are usually implemented at specialized

Hindawi Occupational erapy International Volume 2023, Article ID 6466836, 8 pages https://doi.org/10.1155/2023/6466836

schools [7]. Due to the fact that the problem is global, there are quite a lot of options for such training. Various cognitive behav- ioral practices are very often used to teach interpersonal rela- tionships [8]. For example, there are real-time play therapy techniques: each participant must build a dialogue and learn to solve various problems together with another person or a group of people under the supervision of a teacher or psychol- ogist [9]. Cognitive therapy options using various mobile appli- cations are also being considered. These can be simple games with cards that help children communicate with others and by choosing one or another card, to show their emotions and desires. This makes it easier to understand the needs of children [10]. There are applications that simulate certain situations, teach reactions, and, through repetition, reinforce the acquired skill. For example, there are mobile phone applications that match a word to an object or colour [11]. Currently, consider- ation is being given to introducing a virtual universe using com- puter technology. This makes it possible to expand the variability of tasks and enhance visual fixation during trainings [12]. Despite the positive dynamics and all possible options for the socialization of children with intellectual disabilities, it should be borne in mind that most studies have certain gaps. Many of them require additional research in various focus groups to obtain new results [13]. The problem of the culture of relationships among people with intellectual disabilities is quite relevant. Their inability to build social relationships reduces the quality of life and prevents them from becoming full-fledged members of society. Therefore, new training and psychological assistance programs are constantly being devel- oped in the world. This not only provides an opportunity for socialization but also helps to change the attitude of society towards people with impaired intellectual functions. More research is required to understand the effectiveness of the impact of cognitive techniques on interpersonal culture. It is necessary to introduce similar experimental methods in schools and boarding schools for children with intellectual disabilities.

2. Literature Review

Scientists from Sydney (Australia) conducted research on impaired social interaction skills among children with intel- lectual disabilities. They concluded that the main problem lies in the impairment of the neuropsychological profile as such children often have affective and comorbid disorders. This leads to impaired memory, learning, and expression of one’s own thoughts and emotions. In most cases, atten- tion deficit hyperactivity disorder occurs. Due to this, com- munication becomes difficult. It was also concluded that there are very few methods for helping such children and the best choice is cognitive behavioral therapy. However, the data on the effectiveness of the influence turned out to be rather controversial [14]. There was an experiment con- ducted in the UK to introduce cognitive programs to help people with congenital disabilities. The concept was to learn to distinguish between behaviors, thoughts, feelings, and emotions and to relate them to different situations. It was a randomized study involving people with mild to moderate mental retardation. These training sessions significantly improved understanding of one’s own emotions, as well as

the ability to respond correctly to various life situations [15]. Swedish researchers considered the impact of cognitive group training on the socialization of children with autism spectrum disorders. As part of the experiment, the effective- ness of this method has been confirmed [16]. Studies by South Korean researchers on the issue of socialization and improvement of communication skills among people with intellectual disabilities also confirmed their effectiveness. The methodology aimed to teach people the rules of com- munication, etiquette, and cultural characteristics of the region. An intragroup study demonstrated a fairly high effi- ciency [17]. In Victoria (Australia), researchers considered the effects of cognitive behavioral therapy on the social skills of children and adolescents. A randomized experiment, which confirmed the positive impact of behavioral interven- tions on children with socialization deficits, was carried out [18]. Scientists from Maribor (Slovenia) considered play intervention for children with developmental disabilities. All games were built on the development of cognitive skills, fine motor skills, social skills (adequate interpretation of emotions), and expansion of vocabulary. All games were played in a digital environment. The experiment studied children with autism and attention deficit disorder. How- ever, the results were controversial. While the serious play method reduced anxiety and stress indicators and contrib- uted to the recognition and management of emotions, clini- cal evidence of the benefits of this strategy was not revealed [19]. In Ireland, a strategy for the development of communi- cation and language skills in children with Down syndrome was considered. The study said that the development of the social abilities in these children should start at a very early age. This is associated with the fact that contact and commu- nication with people around them affects the level of com- munication skills of people with a similar disorder. Thus, it was concluded that social skills should be improved not only by the teacher but also by the parents or guardians. This requires that parents are taught to find contact with their children [20]. Indian researchers have studied the preoccu- pation with activities in children with intellectual disabilities. The experiment involved two focus groups, one of which consisted of absolutely healthy children, and the second one included children with various types of intellectual dis- abilities. The researchers let the children play in the class- room with ordinary toys (construction kits, figurines, cars, dolls) and watched the process. As a result, they noticed that the immersion and enthusiasm of children with mental dis- abilities in the game is much higher than that of healthy chil- dren. This should be taken into account as a psychological feature of children with intellectual disabilities in order to properly select a correction program [21]. Based on the data obtained, some conclusions can be drawn about teaching communication skills to people with intellectual disabilities. First, it should be borne in mind that the main socialization problems mainly occur due to the presence of comorbid and affective disorders. It should be taken into account that ear- lier development will determine the effectiveness of the selected strategy. Secondly, there is a need to choose the right approach for these children. One of the best options is cognitive therapy. There are a lot of cognitive therapy

2 Occupational Therapy International

options: from individual sessions with a specialist to ready- made mobile applications and computer games. Thirdly, it is worth noting that most of the experiments are randomized studies. Therefore, precisely accurate results cannot be con- firmed and such strategies require further research.

2.1. Setting Objectives. The motivation is to conduct more studies on the impact of cognitive therapy on the culture of interpersonal relationships among children with intellec- tual disabilities to understand the degree of effectiveness. Cognitive therapy has not yet been completely studied, and more research is needed. In the course of the study, it is also worth finding out in which areas this technique can be applied and how appropriate it is. The main goal was to study the influence of cognitive behavioral therapy on the socialization of children with intellectual disabilities aged 11-13. A randomized study was conducted at specialized (correctional) boarding school No. 7 and Zhanuya boarding school. A total of 350 children took part in the study. The objectives included the introduction of cognitive therapy in the education of children with intellectual disabilities. Also, to clarify the degree of socialization problems and test the effectiveness, specialized testing was used to assess commu- nicative competence. This test was implemented to evaluate the effectiveness of the integrated methodology. Further prospects of using the cognitive therapy strategy were also assessed. Specialized testing was used to analyze progress in the study. This was carried out and evaluated by a special- ist in psychology.

3. Methods

3.1. Research Design and Sample. A randomized study was conducted to assess the impact of cognitive behavioral therapy on children with intellectual disabilities. This study looked at the uncontrolled before-after design. There was no control group in the study. The experiment involved students of specialized boarding school No. 7 and Zhanuya boarding school (Almaty). These were 5-7 grade students. The age of the respondents was 11-13 years old. The experimental group consisted of 350 participants. There were 180 boys and 170 girls among the respondents. The objectives of the experiment were to introduce cognitive therapy into the education of children with intellectual dis- abilities. A specialized test to assess communicative compe- tence was also used to clarify the extent of socialization problems and to verify effectiveness. This questionnaire was administered to evaluate the effectiveness of the com- prehensive methodology. Further perspectives of the cogni- tive therapy strategy were also evaluated. Specialized testing was used to analyze the progress of the study. It was conducted and evaluated by a specialist in psychology. Outside the study, the children attended school in accor- dance with their timetable. No additional psychological therapy was performed.

3.2. Experiment. Prior to the experiment, all respondents were tested to evaluate their communicative and social com- petence. This is a multifactorial questionnaire that makes it

possible to assess the position of the individual in society and make a probable forecast of their further social activity. It includes 100 questions and the assessment of such criteria as factor A (openness, sociability), factor D (assessment of cheerfulness), factor K (desire to work in a team), factor P (propensity for antisocial behavior), factor M (independence and independence of other people’s opinions), factor N (self- control), and factor V (developed logical thinking). Each indicator is assessed on a 20-point scale except for factor P that is evaluated on a 40-point scale. Psychometric proper- ties of this test include reliability (0.94-0.90), validity (0.85- 0.8), and discriminability (0.33) [22]. The participants were tested at the beginning, in the middle, and at the end of the study. The experiment took place over four months. The therapy sessions were carried out 3 times a week. Each session lasted 3 hours. All training sessions were conducted under the guidance of a psychologist and a supervisor. Each of the three sessions that were carried out during the week had its own specifics. As part of the first type of training, col- lective therapy aimed at identifying the weaknesses of the interaction and their correction was carried out. This involved group sessions. Thus, a small group of children was selected to find a way out of a simulated situation through interaction with each other and teamwork. All respondents had to solve at least one case problem, after which they had to tell what emotions they experienced and analyze them [23]. The second type of activity was held in a training format. The children were shown standard models of interaction between people in various situations and had to repeat them to consolidate the material while acting out the scenario with each other [9]. The third type of session aimed to teach children to build a dialogue with each other by expressing their emotions and requests with the simula- tion of certain situations [24]. During these sessions, the respondents were divided into pairs. At the end of each month of the experiments, groups and pairs were disbanded and new ones were created to increase the level of adaptation of the respondents.

3.3. Statistical Processing and Data Analysis. The analysis was carried out using a questionnaire on communicative and social competence. It is a multifactor questionnaire which includes a measurement on eight criteria. The test itself consists of 100 questions. Seven of the criteria were assessed on a scale of 0 to 20 points, and only factor P (propensity for antisocial behavior) was assessed on a 40- point scale. Psychometric properties of this test include reliability (0.94-0.90), validity (0.85-0.8), and discriminabil- ity (0.33) The multifactor questionnaire is validated by Nikolai Petrovich Fetiskin (vice president of the Interna- tional Academy of Psychological Sciences). This question- naire was administered to all respondents three times: before the experiment, in the middle (interim testing), and at the end of the study.

To process the data of this study, a specialized program for statistical analysis SPSS 26.0 was used. The results were interpreted and visualized with the help of the Microsoft Excel 2019 software package. Student’s t-test was used to compare the effectiveness of the proposed teaching approach.

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To compare the mean result of the initial test with the results of the posttest to identify significant differences in the learn- ing process. The level of significance was set at (p ≤ 0:05). 95% confidence intervals (CI) were calculated for median analysis.

3.4. Research Limitations. It should be noted that the degree of intellectual disability was not taken into account; all children were treated equally during the experiment. How- ever, it should be taken into account that due to the vary- ing degrees of intellectual disability, this therapy may not affect the participants in the same way. Also, the disease, which led to intellectual impairment, and its specificity were not taken into account. The experiment involved children with various pathologies. It should be noted that this is a completely randomized trial, the purpose of which is to determine the impact of cognitive behavioral therapy on the socialization of children with intellectual disabilities and teaching them communication skills. The results of the study will be considered in terms of the arithmetic mean of the sample. All these factors should be taken into account when considering the results of the experiment. It should also be noted that the study was not carried out after the experiment had ended. It is therefore not possible to say with certainty that the result remains at a static level.

3.5. Ethical Issues. Such experiments require the consent of the respondents. Due to the fact that the prospective par- ticipants have not yet reached the age of majority, the official decision can only be made by the persons respon- sible for them. Parents and guardians were informed ver- bally and in writing about the format of the experiment and all its details. After that, they gave their written con- sent for their children’s participation in the study. The children gave their verbal consent to take part in the study after having been told about the essence of the experiment.

4. Results

Before the experiment began, a communicative competence test was carried out. All the results of the respondents have an average value for the study group and are described as points and percentages. The preliminary result for factor A (openness and sociability) was 8 points (40%)—the lowest indicator of the average level of sociability. The result for factor D (cheerfulness) was 13 points (65%). The indicator of factor K (teamwork) turned out to be rather low -5 points (25%). Factor P (propensity for antisocial behavior) result was equal to 12 points out of 40 possible (30%). The score for factor M (independent decision-making) turned out to be rather low -6 points (30%). Factor N (self-control) also showed a low value of -5 points out of 20 possible (25%). Factor V (logical thinking) was equal to 4 points (20%). The result of factor C (emotional stability) is 6 points (30%), which is also a rather low indicator. The result is shown in Table 1.

Intermediate testing was carried out 2 months after the start of the experiment. The factor A results (openness and sociability) were 11 points (55%). This indicates an increase in sociability in the study group. In turn, factor D (cheerful- ness) changed insignificantly; it increased by 1 point (70%). The factor K indicator (teamwork) increased by 3 points; thus, it was equal to 8 (40%). In two months, this indicator moved from the low level to the average level. Factor P (pro- pensity for antisocial behavior) decreased to 10 points out of 40 possible (25%). The result of factor M (independent decision-making) increased to 9 points (45%). Factor N increased by two points and was equal to 7 points (35%). Logical thinking (factor V) also increased by 2 points and was equal to 6 points out of 20 possible (30%). Emotional stability (factor C) changed its value in a positive direction -8 points (40%). The result of intermediate testing is shown in Table 2.

At the end of the experiment, the final test was carried out. According to its results, factor A (openness and sociabil- ity) reached 13 points (65%). The factor D result (cheerful- ness) was 15 points (75%)—the upper limit of the average level. Factor K (teamwork) amounted to 10 points (50%). The results of factor P (propensity to antisocial behavior) were equal to 8 points out of 40 (20%). The factor M indica- tor (independent decision-making) was 11 points (55%). The factor N indicator (self-control) was equal to 9 points out of 20 possible (45%). The result of factor V (logical thinking) was 8 points (40%). Factor C (emotional stability) reached 10 points (50%).

The result of the final testing is shown in Table 3. After the final test was calculated, it was compared with

the preliminary results using Student’s t-tests (Table 4). The significance level of A, K, and M is 0.008 (p ≤ 0:01). This result demonstrates the exclusion of the null hypothesis and acceptance of the alternative hypothesis. That is, it shows that there is a sufficient statistical difference between the “before” and “after” figures. The significance level of fac- tor D is 0.005 (p ≤ 0:01). The significance level of factors P, V, and C is equated to 0.011 (p ≤ 0:05). And factor N has a significance level of 0.001 (p ≤ 0:001). It follows that all cri- teria underwent statistically significant changes in the course of the experiment.

Based on the data above, we can conclude that cognitive therapy sessions have a positive effect on the development of social skills in children with intellectual disabilities. This is evidenced by an increase in the indicators of teamwork, independent decision-making, self-control, logical thinking, and emotional stability. The criteria, such as “cheerfulness,” “openness,” and “sociability” also improved. It is worth not- ing that the factor of the propensity for antisocial behavior decreased due to the sessions of cognitive therapy. Also, changes in the behavior of the students who took part in the experiment began were noticed by their teachers. According to one of the supervisors, the children have really become more open in communication with each other and teachers, in particular. They are more actively involved in learning and open up to everything new with great interest. It is also worth noting that their academic performance has improved.

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However, it should be considered that the study is completely randomized and was conducted in a large focus group. Also, the disease that provoked intellectual disability

and its degree were not taken into account. Therefore, the absolute reliability of the results cannot be stated. In the future, additional research may be required.

Table 2: Intermediate results.

Name of factor Points Standard deviation %

Factor A (openness, sociability) 11 points 1.1 55%

Factor D (cheerfulness) 14 points 1.4 70%

Factor K (teamwork) 8 points 1.2 40%

Factor P (propensity for antisocial behavior) 10 points 1.3 25%

Factor M (independent decision-making) 9 points 1.0 45%

Factor N (self-control) 7 points 0.8 35%

Factor V (logical thinking) 6 points 1.1 30%

Factor C (emotional stability) 8 points 1.0 40%

Table 3: Final results.

Name of factor Points Standard deviation %

Factor A (openness, sociability) 13 points 0.7 65%

Factor D (cheerfulness) 15 points 0.6 75%

Factor K (teamwork) 10 points 1.1 50%

Factor P (propensity for antisocial behavior) 8 points 0.9 20%

Factor M (independent decision-making) 11 points 1.0 55%

Factor N (self-control) 9 points 0.7 45%

Factor V (logical thinking) 8 points 0.6 40%

Factor C (emotional stability) 10 points 0.9 50%

Table 1: Preliminary results.

Name of factor Points Standard deviation %

Factor A (openness, sociability) 8 points 1.3 40%

Factor D (cheerfulness) 13 points 1.6 65%

Factor K (teamwork) 5 points 1.2 25%

Factor P (propensity for antisocial behavior) 12 points 1.8 30%

Factor M (independent decision-making) 6 points 1.5 30%

Factor N (self-control) 5 points 1.1 25%

Factor V (logical thinking) 4 points 1.2 20%

Factor C (emotional stability) 6 points 1.4 30%

Table 4: Comparison of the results.

Scales Preliminary results. Final results Empirical value Significance level (p value)

Factor A 8 ± 0:141 13 ± 0:283 -22.361 0.008∗∗

Factor D 13 ± 0:141 15 ± 0:141 -14.142 0.005∗∗

Factor K 5 ± 0:283 10 ± 0:141 -22.361 0.008∗∗

Factor P 12 ± 0:141 8 ± 0:283 17.889 0.011∗

Factor M 6 ± 0:283 11 ± 0:141 -22.361 0.008∗∗

Factor N 5 ± 0:141 9 ± 0:141 -28.284 0.001∗∗∗

Factor V 4 ± 0:141 8 ± 0:283 -17.889 0.011∗

Factor C 6 ± 0:283 10 ± 0:141 -17.889 0.011∗

∗p < 0:05; ∗∗p < 0:01; ∗∗∗p < 0:001.

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5. Discussion

In 2019, a study on the thinking and behavior of chil- dren with intellectual disabilities was conducted in the Netherlands. The emphasis was placed on the assump- tion of the flexibility of behavior and personality factors. A randomized trial was conducted. The results showed that children with intellectual disabilities have more fixed think- ing in terms of emotions and behavior. This suggests that teaching positive attitudes towards the development of the emotional sphere can be used to correct behavior and improve mental state [25]. It is impossible to fully compare this study with the present paper. This is due to the fact that the experiment, which was carried out in the Nether- lands, does not involve cognitive therapy. However, the result obtained in the course of the Dutch research describes and confirms the positive dynamics that were obtained during the experiment described above in this study. In 2018, Canadian scientists conducted an experi- ment and studied the effect of cognitive behavioral therapy on the behavior of children with autism. The study was randomized. As a result, the scientists concluded that the indicators of emotional state and socialization skills improved in the focus group [26]. Comparing the results obtained by Canadian scientists with the results of this study, we can say that in both cases, there is a positive trend. The only difference is that in Canada, the focus group consisted exclusively of children with autism while this study looked at intellectual spectrum disorders more extensively. In 2018, there was an American study of the effect of cognitive therapy on children with self-regulation problems, namely, with social, executive dysfunction, and emotional spectrum disorders. The results proved the effec- tiveness of the impact of cognitive behavioral therapy on these areas of life [27]. This also confirms the feasibility of the application of this type of therapy to solve the prob- lem of socialization in children with intellectual disabilities. Scientists from the UK have studied the effect of cognitive behavioral therapy on people with intellectual disabilities. They conducted a randomized trial. The results showed improved cognitive functions and improved quality of life [28]. The experiment by British researchers has confirmed the results of this study. Joint developments of scientists from China and the United States were based on studying the effectiveness of the use of cognitive therapy for resis- tance to various social situations in people with neurocog- nitive disorders. The results were ambiguous. Resistance to various social factors increased, but this did not affect neuropsychiatric behavioral symptoms [29]. However, the authors of this study argue that there could be various inac- curacies in the course of the experiment; therefore, absolute results cannot be claimed. Due to the fact that most studies in this area have positive dynamics, we can talk about con- firmation of the effectiveness of this strategy, but it is also worth considering all the nuances associated with the design and conduct of these experiments. It is worth men- tioning the fact that most studies are randomized; as a result of this, there may be certain gaps. It is impossible to identify the criteria for the effectiveness of the influence

of cognitive therapy on people with certain disabilities as specific diseases and the degree of their development are not taken into account in most cases.

The results obtained can be explained in terms of the fact that cognitive therapy is a learning factor. During the ses- sions, people work out new patterns of behavior reinforcing them by repetition. As people with various intellectual dis- abilities are able to learn the expression of various emotions and their manifestation in different situations, we can talk about an increase in acquired social skills.

The results described in this study are also randomized and cannot claim absolute accuracy; therefore, additional research is required in this area. During the comparison of this work with other articles, no fully similar methodology was found. It follows that this strategy may be a unique development.

The application of the knowledge gained gives us the understanding of the effectiveness of cognitive therapy in the development of a culture of interpersonal relationships in people with intellectual disabilities. This strategy can be used in the context of learning and developing social skills. This provides a prospect for new discoveries in the field of interpersonal relationships, as well as changing and improv- ing the cognitive techniques aimed at people with a narrower specificity of intellectual disabilities.

However, it must be kept in mind that the degree of mental retardation was not taken into account; all children were treated equally in the experiment. Because of the vary- ing degrees of mental retardation, the therapy may not affect the participants in the same way. The illness that led to men- tal retardation and its specifics were also not taken into account. Children with different pathologies participated in the experiment. This is a fully randomized study that aims to determine the effects of cognitive behavioral therapy on the socialization of children with intellectual disabilities and their learning of social skills. It should also be noted that the study was not carried out after the experiment had ended. It is therefore not possible to say with certainty that the result remains at a static level.

6. Conclusions

The results of this study showed that the use of cognitive therapy for the development of a culture of interpersonal relationships in children with intellectual disabilities has proven to be effective. This conclusion can be made based on the improvement of the indicators of self-control, team- work, independent decision-making, logical thinking, and emotional stability. The indicators moved from the low level to the average one, which indicates an improvement in socialization skills. It is also worth noting a slight increase in the indicators of cheerfulness and sociability. The factor of antisocial behavior, in turn, decreased, which indicates a positive trend in the study. This leads to the conclusion that it is advisable to use cognitive therapy for the socialization of children with intellectual disabilities.

The scientific value of this experiment lies in the applica- bility of the results for further research and the improvement of the methods of cognitive therapy in this area. It should be

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borne in mind that the study was based on a randomized sample method; therefore, this strategy requires additional experiments to obtain more accurate results. However, the method of cognitive therapy can be used to develop a culture of interpersonal relationships in people with intellectual dis- abilities. It should be used in schools and specialized institu- tions for children with intellectual disabilities. Also, the method of cognitive therapy can be used for preschool chil- dren with intellectual disabilities; however, it should be con- sidered that this strategy may need to be adapted for this age group. Cognitive therapy can also help adults with intellec- tual disabilities socialize. Therefore, this strategy can be applied in private psychological practice.

When assessing the prospects for further research, it is worth pointing out the gaps in this research. The disadvan- tage is a rather large focus group of respondents, which does not give an absolute specificity of the results. It should be noted that due to the large number of participants, all results are considered in the mean value system. Also, intellectual disorders and the degree of their severity were not consid- ered and were not taken into account. Standard cognitive therapy methods were chosen to promote socialization. Therefore, further research should examine the impact of such a strategy on a small group of respondents. Cognitive therapy to specific cases of intellectual disability, such as Down syndrome or autism, can also be applied. To obtain more accurate results, the sample should include respon- dents with the same severity of specific diseases. Other methods of cognitive therapy can also be applied. The results of such studies will make it possible to correct various cogni- tive methods in accordance with the degree and form of intellectual disabilities. This will contribute to the develop- ment of an individual approach and increased efficiency.

Data Availability

The data will be available on request.

Conflicts of Interest

The authors declare that there is no conflict of interest regarding the publication of this paper.

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Journal of Autism and Developmental Disorders (2024) 54:3316–3335 https://doi.org/10.1007/s10803-023-06063-x

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ORIGINAL PAPER

Cognitive‑Based Interventions for Improving Psychological Health and Well‑Being for Parents of Children with Developmental Disabilities: A Systematic Review and Meta‑analysis

Sini Li1,2 · Yijing Yong3 · Yamin Li2 · Jianhe Li4 · Jiao Xie2,5

Accepted: 6 July 2023 / Published online: 5 September 2023 © The Author(s) 2023

Abstract This review aims to systematically summarize existing evidence to determine the effectiveness of cognitive-based interven- tions (CBIs) on psychological health and well-being among parents of children with developmental disabilities (DD). Six databases were searched to identify eligible randomized controlled trials (RCTs) from their inception to April 2023. The revised Cochrane Risk of Bias tool for RCTs was applied to assess the risk of bias and the certainty of evidence was evalu- ated using the Grading of Recommendation, Assessment, Development and Evaluation. Meta-analyses were conducted using a random-effects model. Twenty-five RCTs involving 1915 participants were identified. The results indicated that CBIs reduced parental stress levels (Hedges’ g = − 0.69), depressive symptoms (g = − 0.95), anxiety levels (g = − 0.78), and parental distress (g = − 0.29), and improved parental well-being (g = 0.62) and parent‒child relationships (g = 0.43) postintervention compared with the active/inactive control groups. Subgroup analysis of the effectiveness of interventions using mindfulness-based interventions and cognitive behavioural therapy showed positive effects. The favourable interven- tion duration and participant targets were also identified in this review. Furthermore, the effects of CBIs were impacted by the different types of DD among the children. This review highlighted the positive effects of CBIs on parental stress levels, depressive symptoms, anxiety levels, parental distress levels, parental well-being levels, and parent‒child relationships. Future well-designed RCTs are needed to further investigate the effects of MBIs and CBT interventions on children with DD and their parents, as well as the factors and mechanisms of action affecting the efficacy of these interventions.

Keywords Developmental disabilities · Parents · Cognitive behavioural therapy · Mindfulness · Meta-analysis

Introduction

Developmental disorders (DD) are a set of lifelong disor- ders (e.g., attention-deficit/hyperactivity disorder [ADHD], autism spectrum disorder [ASD], cerebral palsy [CP], fragile X syndrome, developmental delay, intellectual dis- abilities [ID], learning disorders, language disorders, and Tourette syndrome) that originate in childhood and cause severe impairment in daily function in different domains (e.g., physical, learning, language, emotional or behav- ioural function) (Holm, 1989; Prevention, 2022). Previous nationwide surveys in the United States discovered that the prevalence of DD has risen from 12.84 to 17.76% in the last 20 years (Boyle et al., 2011; Zablotsky et al., 2019). Given this growth, substantial care needs, support, and accessibil- ity of resources and interventions are typically required for children with DD and their families, enhancing long-term

* Jianhe Li [email protected]

* Jiao Xie [email protected]

1 The Nethersole School of Nursing, Faculty of Medicine, The Chinese University of Hong Kong, Hong Kong, China

2 Clinical Nursing Teaching and Research Section, The Second Xiangya Hospital, Central South University, 139 Renmin Middle Road, Changsha 410011, China

3 Cognition and Human Behaviour Key Laboratory of Hunan Province, Hunan Normal University, Changsha, China

4 Department of Pharmacy, The Second Xiangya Hospital, Central South University, 139 Renmin Middle Road, Changsha 410011, China

5 Department of Otolaryngology-Head and Neck Surgery, The Second Xiangya Hospital, Central South University, 139 Renmin Middle Road, Changsha 410011, Hunan, China

3317Journal of Autism and Developmental Disorders (2024) 54:3316–3335

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outcomes for those children and families (Baio et al., 2018; Vohra et al., 2014).

Parenting a child with DD is exhausting, and it imposes many challenges on parents because they need to invest considerable time, energy, and money to support and nur- ture their child’s healthy development (Resch et al., 2010). Due to the specialized care needs of these children, par- ents have observed amplified levels of emotional distress (Robinson et al., 2018), also known as stress (Osmančević Katkić et al., 2017), anxiety (Bujnowska et al., 2019), and depression (Scherer et al., 2019), as well as impairments in other health outcomes, such as physical function (Cantwell et al., 2014), social function (Ali et al., 2012), and general well-being (Baker et al., 2005) or quality of life (Arora et al., 2020). Concerningly, these psychological issues can have a detrimental impact on parental well-being and parenting practice (Neece et al., 2012), resulting in poor child, parent, and family outcomes in families with children with DD due to bidirectional relationships between parent emotion and child outcomes (Woodman et al., 2015).

To support the parents of children with DD and improve their psychological well-being, a variety of cognitive-based interventions (CBIs), including cognitive-based therapy (CBT), dialectical behavioural therapy (DBT), mindfulness- based intervention (MBIs), acceptance and commitment therapy (ACT), and compassion-focused therapy (CFT), have been recently developed. Based on the concept that thoughts, feelings, and behaviours are interrelated, CBT employs cognitive methods to change maladaptive beliefs and cognitive distortions as well as modify problematic behavioural patterns that maintain stress/distress (Beck & Haigh, 2014). Moreover, MBIs, which include mindfulness- based stress reduction (MBSR) and mindfulness-based cog- nitive therapy (MBCT), can be defined as the practice of mindfulness that improves participants’ attention/awareness of the present moment in a nonjudgmental manner, allow- ing them to better cope with life stressors and enhance their general well-being (Kabat-Zinn, 2003). The first applica- tion of these ideas to psychotherapy was developed by Dr. Jon Kabat-Zinn in the 1970s by first applying MBSR in the Western world (Kabat-Zinn & Hanh, 2009). The principle of MBSR has been further adapted in MBCT, which combines the mindfulness practices of MBSR with the concepts of CBT to treat the process of negative thinking and feeling rather than the content of psychological symptoms (Chad- wick et al., 2016), or ACT, which aims to improve affective symptoms by promoting psychological flexibility, identify- ing personal values, and managing commitments to make adjustments (Hayes & Strosahl, 2005; Hayes et al., 1999). In addition, as part of third-wave CBT, DBT combines various strategies (e.g., mindfulness, emotion regulation, awareness, and acceptance) that enable clients to utilize new skills/strat- egies to build lives that they feel worthwhile. Furthermore,

as newly developed CBI, CFT incorporates mindfulness to assist in enabling mental and emotional recovery by encour- aging participants to be compassionate towards themselves and others (Gilbert, 2009; Khoshvaght et al., 2021).

These CBIs have reported promising results in lessen- ing stress, depressive symptoms, and anxiety across parents of children with special needs (Bourke-Taylor et al., 2021; Parmar et al., 2021), further confirming their ability to help enhance the psychological well-being of parents of children with DD. Previous systematic reviews and meta-analyses reported significant improvements in the psychosocial out- comes of caregivers of children with ASD with CBIs (Yu et al., 2019). This finding was further confirmed by Bourke- Taylor et al. (2021), who also revealed the positive effects of CBT on parental stress and mental health for parents of children with DD. Several systematic reviews demonstrated that MBIs and/or ACT were superior to the control group regarding postintervention mental health and subjective well-being results for parents of children with different types of DD (Chua & Shorey, 2022; Hartley et al., 2019; Juvin et al., 2022; Lee et al., 2022; Yu et al., 2019).

While the reviews mentioned above offer preliminary evi- dence on the effectiveness of CBIs for parents of children with DD, these studies contain significant methodologi- cal limitations. First, the included studies in these reviews used a wide range of study designs, including single-group, pre- and post-test studies, randomized controlled trials, and nonrandomized controlled trials (Hartley et al., 2019; Juvin et al., 2022; Lee et al., 2022; Yu et al., 2019). Therefore, those syntheses might provide low-quality evidence with a significant risk of bias and unreliable outcomes. In addi- tion, the majority of previous reviews included a limited number of studies and small sample sizes (lower than 1,000 participants), making it difficult to draw solid conclusions (Cachia et al., 2016; Hartley et al., 2019; Juvin et al., 2022; Lee et al., 2022; Osborn et al., 2021). Furthermore, the study samples were very heterogeneous, including children and adults with DD (Hartley et al., 2019; Juvin et al., 2022), and did not analyse outcomes separately for parents of children with different types of DD (Bourke-Taylor et al., 2021; Chua & Shorey, 2022; Osborn et al., 2021).

Considering the heavy caregiver burden of those parents, the limitations of the systematic and meta-analysis demon- strated above, and the potential benefits of CBIs for parents of children with DD to improve their mental health, it is paramount to synthesize the existing evidence on the use of CBIs to enhance parents’ psychological well-being in while caring for children with DD to drive future evidence-based research and practice. Moreover, it is still inconclusive about the best effective approach and optimal characteristics of CBIs for improving those parents’ health outcomes. There- fore, this review aimed to comprehensively summarize the effects of CBI with respect to psychological problems and

3318 Journal of Autism and Developmental Disorders (2024) 54:3316–3335

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well-being among parents of children with DD, when com- pared with active/inactive controls, and investigate the opti- mal features of the effective interventions found.

Methods

This review was registered with PROSERO registration number CRD42022382502 and conducted based on the Pre- ferred Reporting Items for Systematic Reviews and Meta- Analysis statement (PRISMA) (Moher et al., 2009). Two reviewers (SNL and YJY) independently conducted certain phases of systematic review, including study selection, data extraction, and quality appraisals. When confronted with a conflict, two reviewers either discussed or consulted with the third reviewer (YML) to achieve an agreement.

Research Strategy

Six English databases, including PubMed, Embase (Ovid), PsycINFO, CINAHL, Cochrane Central Register, and ProQuest, were systematically searched from inception to December 2022. The PICOS (population, intervention, com- parisons, outcomes, study) framework listed in Appendix S1 was used to select MeSH terms and keywords, includ- ing types of participants (e.g., caregiver*, parent*, mother*, father*, maternal*, famil*), types of diagnoses (e.g., devel- opmental disabilities, autism spectrum disorder, attention deficit disorder with hyperactivity, fragile x syndrome, Down syndrome), types of interventions (e.g., cognitive behavio?ral therap*, acceptance and commitment therapy, mindful, mindfulness-based, dialectical behavioural ther- apy, metacognitive), and types of psychosocial outcomes (e.g., stress, anxiety, depressi*, parenting distress). The search strategy was designed for PubMed and then adapted for other databases. The research strategies and results for all the databases are listed in Supplementary Tables 2–7. Moreover, the reference lists of pertinent publications and internet search engines, such as Google Scholar, were manu- ally searched for additional relevant articles.

Inclusion and Exclusion Criteria

Studies were included when they met the following criteria:

(1) Populations: The participants were parents (aged > 18) of children (aged ≤ 18) diagnosed with DD (accord- ing to the World Health Organization (Almeida et al., 2020), the diagnoses for DD include a diverse group of conditions characterized by impairments in physi- cal ability, learning, language and/or behaviour, such as autism spectrum disorder [ASD], attention-deficit/

hyperactivity disorder [ADHD], fragile X syndrome, Down syndrome, intellectual disabilities).

(2) Interventions: The studies explicitly and independently referred to CBIs, including CBT, MBIs, ACT, DBT, or self-compassion therapy, in their description of the primary part of the intervention.

(3) Comparison: The CBIs were compared to both active (e.g., attention care) and inactive (e.g., treat as usual [TAU], standard care, waitlist, no treatment, placebo) control groups.

(4) Outcomes: The parental psychological outcomes (including emotional distress, stress, depression, and anxiety) were measured by valid instruments at least at the time of postintervention.

(5) Study design: Only randomized controlled trials (RCTs) published in peer-reviewed journals and (6) only stud- ies in English were eligible.

Studies were excluded when (1) the primary outcomes of interventions were focused on children with DD (e.g., child-focused or parent-mediated interventions) rather than their parents; (2) multicomponent interventions (such as combined CBIs with behavioural training) were considered the major component in the intervention group; (3) the group sample size for those receiving treatment was lower than five; and (4) full-text or final results were unavailable (e.g., conference abstracts and protocols).

Study Selection

To check for duplicate publications, all retrieved records were loaded into Covidence software. The abstracts and titles were separately reviewed by two reviewers (SNL and YJY) in accordance with the eligibility criteria. Eligible arti- cles were eventually identified after examining the full texts of possibly relevant research.

Data Extraction

A self-developed data extraction form was applied based on the Cochrane data collection form for RCT review (Sam- bunjak D et al., 2017). The form was piloted on five ran- domly selected eligible articles and revised accordingly. Two reviewers extracted all necessary data independently, and the following information was collected: (1) the basic informa- tion of the studies (including the last names of the authors, the year of publication, and the locations, study designs, and sample sizes); (2) participant characteristics (including the age range and the sex proportion for the children with DD and their parents); (3) characteristics of the interventions (including the types of cognitive-based interventions and the comparators, intervention contexts, research formats, dos- ages, durations, and lengths of follow-ups); and (4) outcome

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measures (including measurement tools, assessment time- point(s), and outcomes [effect sizes]). Two reviewers (SNL and YJY) extracted data individually, and any differences of opinion were resolved by discussion or contact with the third reviewer (JHL).

The first author contacted the corresponding authors to provide additional information if the information provided was not sufficiently detailed (such as using figures to report the outcomes and not revealing the specific data in the arti- cle). When authors did not reply after being contacted twice through email a month apart, the articles were removed from consideration.

Quality Appraisal

The revised Cochrane Risk of Bias tool (RoB2) for RCTs (Sterne et al., 2019) was applied to assess the quality of each included trial. Any disagreements between critical apprais- ers were resolved by discussion and consultation with the third reviewer. Five categories of bias are evaluated by the RoB: selection (random sequence generation and allocation concealment), performance (blinding of participants and personnel), attrition (incomplete outcome data), detection (blinding of outcome assessment), and reporting bias (selec- tive reporting). Bias was rated as “low,” “unclear,” or “high” for each domain for each study. The proportion of research that met each quality rating was then calculated.

Certainty of Evidence

A narrative assessment of the degree of certainty of the evi- dence was presented using the Grading of Recommenda- tion, Assessment, Development, and Evaluation (GRADE) system (Balshem et al., 2011). Based on the five GRADE domains—methodological limitations of the studies or bias risk, indirectness, imprecision, consistency, and publica- tion bias—two independent appraisers assessed the degree of certainty of the evidence and categorized it into four cat- egories: high, moderate, low, and very low.

Data Synthesis

R software was applied to assess the statistical meta- analysis by computing the Hedges’ g standard mean dif- ferences (SMDs) with 95% confidence intervals (CIs) for each study. Given the expected variability between studies and outcomes, the random-effects model was chosen to pool the SMDs across the studies. The effect sizes (ESs) were primarily assessed postintervention; specifically, the time-point was less than four weeks after the interven- tion. Based on Cohen’s categories, ESs were classified as small (g = 0.2), medium (g = 0.5), or large (g ≥ 0.8) (Lip- sey & Wilson, 2001). Moreover, only outcomes from at

least two or more studies were pooled in the model for the meta-analysis as well as for the subgroup analyses. The meta-analysis included three major subgroup analyses to investigate whether efficacy varied among intervention approaches, durations (1–8 weeks and over eight weeks), targeted participants (only parent-involved versus parent‒ child dyads), and the types of DD among the children. A minimum of two trials per subgroup were required for the subgroup analyses (Deeks & Altman, 2022). Furthermore, several change scores, such as those associated with the parent‒child relationship, parental well-being, mindful parenting, mindful awareness, and psychological flexibil- ity, were reversed for clarity, so that positive ESs were always associated with positive clinical results.

Heterogeneity was assessed using I2 and Cochrane’s Q. I2 values of 25, 50, and 75% were considered low, moder- ate, and high heterogeneity, respectively. Cochran’s Q is the standard test statistic that reveals systematic between- study differences. Sensitivity analyses were undertaken to explore whether the results were drastically affected by excluding trials with highly disparate ESs. If meta- analyses contained more than 10 studies, the assessments of publication bias were performed by visually evaluating Egger’s statistical test and funnel plots. An asymmetry map highlights likely missing research as a result of pub- lication bias. In the case of Egger’s test, a P value of < 0.05 was used to establish statistical evidence of asymmetry (Egger et al., 1997). Outliers in which the 95% CI was outside the 95% CI of the overall mean ESs on both sides were identified through visual inspection of the forest plots. Furthermore, outliers were kept if deleting them did not significantly impact the results. A narrative synthesis was also conducted for those trials and outcomes that were excluded from the meta-analyses to give a comprehensive picture of the data and to make comparisons between dif- ferent CBIs.

Results

Study Selection

A total of 2511 records were identified. After excluding 786 duplicates, the titles and abstracts of 1725 articles remained for the initial screening, and 76 eligible articles were retained for further full-text screening. No extra articles were identified from the search engines or reference lists. Finally, a total of 25 RCTs were selected for inclusion in this review. Figure 1 depicts the PRISMA flow chart of the procedure and the results of the article search and selection. Supplementary Table 8 lists all excluded articles following full-text screening.

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Study Characteristics

Supplementary Table 10 summarizes the main character- istics of the 25 included RCTs. Studies were published between 2006 and 2023, and the majority of studies were developed in the United States (38.10%, n = 8) (Chronis et al., 2006; Dykens et al., 2014; Feinberg et al., 2014; Fer- raioli & Harris, 2013; Hahs et al., 2019; Kuhlthau et al., 2020; Neece, 2014; Schwartzman et  al., 2022). Other

research locations included China (n = 4; (Ho et al., 2021; Liu et al., 2021; Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b)), Australia (n = 2; Whittingham et al., 2022; Wong et al., 2010)), Iran (n = 4; Behbahani et al., 2018; Khosh- vaght et al., 2021; Shareh & Yazdanian, 2023; Sharif et al., 2015)), Spain (n = 2; Lobato et al., 2023; Valero et al., 2022), Italy (Marino et al., 2021), the Netherlands (Siebelink et al., 2018), Nigeria (Onyishi et al., 2023), Turkey (Çiçek Gümüş & Öncel, 2022), and India (Pandya, 2021). All of the studies

Fig. 1 PRISMA Flow Diagram of Study Selection

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3322 Journal of Autism and Developmental Disorders (2024) 54:3316–3335

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were two-arm RCTs with a parallel control design, and the most of studies were pilot RCTs (36%, n = 9) (Ferraioli & Harris, 2013; Hahs et al., 2019; Ho et al., 2021; Kuhlthau et al., 2020; Lobato et al., 2023; Marino et al., 2021; Neece, 2014; Schwartzman et al., 2022; Valero et al., 2022).

Characteristics of Participants The 25 RCTs included 1,915 participants (ntreatment = 970, ncontrol = 945) with sample sizes ranging from 14 (Lobato et al., 2023) to 243 (Dykens et al., 2014). The mean age of the parents ranged from 33.5 (standard deviation [SD]: 7.00) (Feinberg et al., 2014) to 47 (range: 37–60) (Wong et al., 2010). The majority of the participants (85.15%) were female, and eight studies included mothers only. The mean age of children with DD varied from 2.83 (SD: 11.00) (Feinberg et al., 2014) to 13.00 (range: 10–16) (Ho et al., 2021), with boys accounting for the majority (70.72%). Four types of DD were identified among the children in the included studies: ASD (n = 10, 40%), ADHD (n = 8, 32%), CP (n = 2, 8%), ID (n = 1, 4%), and blended DD (n = 4, 16%).

Characteristics of Interventions The following five main approaches were classified: (1) CBT (n = 6 Chronis et al., 2006; Feinberg et al., 2014; Onyishi et al., 2023; Schwartz- man et al., 2022; Sharif et al., 2015; Wong et al., 2010)); (2) MBIs (n = 12; (Behbahani et al., 2018; Dykens et al., 2014; Ferraioli & Harris, 2013; Ho et al., 2021; Kuhlthau et al., 2020; Liu et al., 2021; Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b; C. L. Neece, 2014; Pandya, 2021; Sie- belink et al., 2018; Valero et al., 2022)); (3) ACT (n = 5; Çiçek Gümüş & Öncel, 2022; Hahs et al., 2019; Lobato et al., 2023; Marino et al., 2021; Whittingham et al., 2022)); (4) DBT (n = 1; (Shareh & Yazdanian, 2023); and (5) CFT (n = 1; (Khoshvaght et al., 2021)).

Across the included studies, the duration of CBIs ranged from 2 days (Hahs et al., 2019) to 50 weeks (Pandya, 2021) (mean = 70.08 days, SD = 64.98, median = 56). The aver- age number of CBI sessions ranged from 2 to (Hahs et al., 2019) to 24 sessions (mean = 8.4, SD = 4.05, median = 8), except for a study with posted messages (Pandya, 2021). Seven studies adopted the format of a parent‒child dyad (Behbahani et al., 2018; Çiçek Gümüş & Öncel, 2022; Ho et al., 2021; Lo et al., 2017a, 2017b; Neece, 2014; Siebe- link et al., 2018; Valero et al., 2022), and the remaining studies all only involved parents as active participants. Two modes of intervention delivery were identified: face-to-face (n = 21) and online (n = 3), and one study did not report the delivery mode. Moreover, four studies delivered the inter- vention to individuals, while others delivered the interven- tions to groups of participants (n = 21; 84%). The majority

of the investigations disclosed their settings (n = 18), which included in the community (n = 3), hospitals/clinics (n = 6), service/rehabilitation centres (n = 4), schools (n = 2), and internet platforms (n = 3) (Fig. 2).

Characteristics of Control Groups The majority of stud- ies (n = 21) used inactive controls, such as waitlists (n = 13), no treatments (n = 4), and treatments as usual (TAU; n = 4), while the others used active controls, such as child-targeted parenting skills training (n = 2) (Ferraioli & Harris, 2013; Marino et al., 2021), a 1-day mindfulness workshop (Lo et al., 2017a, 2017b), and positive psychotherapy (Dykens et al., 2014).

Outcome Measures Table 1 presents a list of all meas- urement instruments, all of which were valid and reliable questionnaires. Studies used various time-points to obtain their results. One study additionally performed an evaluation at the midpoint of the intervention (Dykens et al., 2014). Over half of the studies (n = 15) used multiple time-points to assess longer-term effects, including short- (less than 3 months) (Behbahani et al., 2018; Khoshvaght et al., 2021; Schwartzman et al., 2022; Sharif et al., 2015), medium- (3 to 5 months) (Chronis et al., 2006; Çiçek Gümüş & Öncel, 2022; Lobato et al., 2023; Onyishi et al., 2023) and long- term effects (6 months or more) (Dykens et al., 2014; Fer- raioli & Harris, 2013; Neece, 2014; Siebelink et al., 2018; Valero et al., 2022; Whittingham et al., 2022; Wong et al., 2010).

Meta‑analyses of CBI Outcomes

The meta-analyses comprised 21 studies in total. Two MBI studies developed by Dykens et al. (2014) and Neece (2014) were excluded from the meta-analyses because the mean and/or SD values could not be obtained from the report. Another MBI study, Pandya (2021), which revealed extraor- dinarily large ESs (Hedges’ g range: 4.93–11.2) for all the long-term results (50 weeks), was considered an outlier for several outcomes (e.g., parental stress, distress, and parent‒ child relationship). Similar to Pandya (2021), the outcomes reported by Çiçek Gümüş and Öncel (2022) were also iden- tified as an outlier, with large ESs ranging from 2.706 to 6.084. The results of the meta-analyses are listed in Table 2.

Parental Stress Sixteen trials with 1015 participants were included to assess the effectiveness of CBIs on paren- tal stress (Behbahani et al., 2018; Chronis et al., 2006; Feinberg et al., 2014; Ferraioli & Harris, 2013; Ho et al., 2021; Liu et al., 2021; Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b; Marino et al., 2021; Onyishi et al., 2023; Schwartzman et al., 2022; Sharif et al., 2015; Valero et al., 2022; Whittingham et al., 2022; Wong et al., 2010). The pooled results showed that CBIs had a significant effect on lessening parental stress when compared with the con- trol group (g = − 0.69, 95% CI [− 1.05, -0.33], P < 0.01,

Fig. 2 a Quality assessment graph about each risk of bias item pre- sented as percentages across all included studies; b Summary of risk of bias assessment of the included randomized controlled trails by Cochrane Collaboration risk of bias tool

◂

3323Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

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H am

ilt on

D ep

re ss

io n

Sc al

e (H

A M

A ; H

am ilt

on , (

19 60

) ( n =

1) Li

u et

 a l.,

(2 02

1) Th

e Pa

tie nt

H ea

lth Q

ue sti

on na

ire -4

K ro

en ke

e t a

l., (2

00 9)

(n =

1) K

uh lth

au e

t a l.,

(2 02

0) D

ist re

ss (n

= 14

) V

is ua

l A na

lo gu

e Sc

al e

(V A

S) –D

ist re

ss L

es ag

e et

 a l.,

(2 01

2) (n

= 1)

K uh

lth au

e t a

l., (2

02 0)

Pa re

nt in

g St

re ss

In de

x— Pa

re nt

al D

ist re

ss S

ub sc

al e

A bi

di n,

(1 99

0) (n

= 1)

H o

et  a

l., (2

02 1)

Pa re

nt in

g St

re ss

In de

x: S

ho rt

Fo rm

— Pa

re nt

al D

ist re

ss S

ub sc

al e

A bi

di n,

(1

99 0)

(n =

9) B

eh ba

ha ni

e t a

l., (2

01 8)

; D yk

en s e

t a l.,

(2 01

4) ; L

iu e

t a l.,

(2 02

1) ; L

o et

 a l.,

(2

01 7a

, b );

M ar

in o

et  a

l., (2

02 1)

; P an

dy a,

(2 02

1) ; S

ch w

ar tz

m an

e t a

l., (2

02 2)

; Va

le ro

e t a

l., (2

02 2)

D ep

re ss

io n

A nx

ie ty

S tre

ss S

ca le

-2 1

Lo vi

bo nd

& L

ov ib

on d,

(1 99

5) (n

= 3)

Ç iç

ek G

üm üş

& Ö

nc el

, ( 20

22 );

Sh ar

eh &

Y az

da ni

an , (

20 23

); Si

eb el

in k

et  a

l.,

(2 01

8) Pa

re nt

al w

el l-b

ei ng

(n =

9) Ry

ff Sc

al es

o f P

sy ch

ol og

ic al

W el

l-B ei

ng —

Sh or

t F or

m (R

yff &

K ey

es ,

(1 99

5) ) (

n = 1)

D yk

en s e

t a l.,

(2 01

4)

G en

er al

H ea

lth Q

ue sti

on na

ire (G

H Q

; G ol

db er

g &

W ill

ia m

s, (1

98 8)

) ( n =

1) Sh

ar if

et  a

l., (2

01 5)

G en

er al

H ea

lth Q

ue sti

on na

ire —

12 (G

H Q

-1 2;

G ol

db er

g &

W ill

ia m

s, (1

98 8)

); (n

= 2)

Lo ba

to e

t a l.,

(2 02

3; W

on g

et  a

l., (2

01 0)

G en

er al

H ea

lth Q

ue sti

on na

ire —

28 (G

H Q

-2 8;

G ol

db er

g, (1

97 8)

) ( n =

1) Fe

rr ai

ol i &

H ar

ris , (

20 13

) W

or ld

H ea

lth O

rg an

iz at

io n-

5 W

el l-b

ei ng

In de

x (W

H O

-5 ; B

ec h

et  a

l., (2

00 3)

) (n

= 3)

H o

et  a

l., (2

02 1)

; L o

et  a

l., (2

01 7a

, b );

Si eb

el in

k et

 a l.,

(2 01

8)

Pe rs

on al

W el

lb ei

ng In

de x

(P W

I; La

u et

 a l.,

(2 00

5) ) (

n = 1)

W hi

tti ng

ha m

e t a

l., (2

02 2)

3324 Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

I2 = 83%; Fig. 3a). Additionally, the robustness of the find- ing was demonstrated by the fact that the pooled results remained unchanged after performing a sensitivity analysis (Supplementary Fig. 1). No publication bias (P = 0.1255) was identified by using Egger’s test and trim-and-fill fun- nel plot (Supplementary Fig. 2).

Depressive Symptoms A total of 12 RCTs including 925 subjects were included to analyse the effects of CBIs on depressive symptoms (Chronis et al., 2006; Feinberg et al., 2014; Hahs et al., 2019; Khoshvaght et al., 2021; Kuhlthau et al., 2020; Liu et al., 2021; Lo et al., 2017a, 2017b; Onyishi et al., 2023; Schwartzman et al., 2022; Shareh & Yazdanian, 2023; Sharif et al., 2015; Whitting- ham et al., 2022). The results indicated a large effect of CBIs on alleviating depressive symptoms compared to the control group (g = − 0.95, 95% CI [− 1.47, − 0.43], P < 0.01, I2 = 90%; Fig. 3b). The robustness of finding was confirmed by sensitivity analysis and trim-and-fill funnel plot, despite Egger’s test revealed potential publication bias (p = 0.0467) (Supplementary Figs. 3, 4).

Anxiety Anxiety was assessed by eight trials with 484 participants (Chronis et al., 2006; Khoshvaght et al., 2021; Kuhlthau et al., 2020; Liu et al., 2021; Onyishi et al., 2023; Schwartzman et al., 2022; Sharif et al., 2015; Whittingham et al., 2022). Compared with the control group, the CBIs identified a significant reduction in anxiety (g = − 0.78, 95% CI [− 1.39, − 0.18], P < 0.01, I2 = 90%; Fig. 3c). The sensitivity analysis confirmed the robustness of the results (Supplementary Fig. 5).

Parental Distress The effectiveness of CBIs on parental distress was evaluated in 11 trials with 838 participants (Behbahani et al., 2018; Ho et al., 2021; Kuhlthau et al., 2020; Liu et al., 2021; Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b; Marino et al., 2021; Schwartzman et al., 2022; Shareh & Yazdanian, 2023; Siebelink et al., 2018; Valero et al., 2022). The results revealed that, in com- parison with the control group, parental distress was sig- nificantly reduced (g = − 0.29, 95% CI [− 0.42, − 0.15], P = 0.44, I2 = 1%; Fig. 3e). Furthermore, following a sen- sitivity analysis in which studies were removed one by one, the pooled results remained constant, confirming the robustness of the finding (Supplementary Fig. 6). The Egg- er’s test (P = 0.1042) and funnel plot also revealed no evi- dence of potential publication bias (Supplementary Fig. 7).

Parental Well-being Eight RCTs involving 426 subjects were used to examine the effects of CBIs on parental well- being (Ferraioli & Harris, 2013; Ho et al., 2021; Lo et al., 2017a, 2017b; Lobato et al., 2023; Sharif et al., 2015; Sie- belink et al., 2018; Whittingham et al., 2022; Wong et al., 2010). The pooled results showed that CBIs had a medium effect on enhancing parental well-being (g = 0.62, 95% CI [0.20, 1.03], P < 0.01, I2 = 69%; Fig. 4a). The sensitivity

Ta bl

e 1

(c on

tin ue

d)

O ut

co m

es M

ea su

re m

en ts

Re fe

re nc

es

Q ua

lit y

of re

la tio

ns hi

p (p

ar en

t– ch

ild ) (

n = 10

) Pa

re nt

in g

St re

ss In

de x—

Pa re

nt –c

hi ld

D ys

fu nc

tio na

l I nt

er ac

tio n

Su bs

ca le

A

bi di

n, (1

99 0)

(n =

1) H

o et

 a l.,

(2 02

1)

Pa re

nt in

g St

re ss

In de

x: S

ho rt

Fo rm

— Pa

re nt

–c hi

ld D

ys fu

nc tio

na l I

nt er

ac tio

n Su

bs ca

le A

bi di

n, (1

99 0)

(n =

9) B

eh ba

ha ni

e t a

l., (2

01 8)

; C hr

on is

e t a

l., (2

00 6)

; L iu

e t a

l., (2

02 1)

; L o

et  a

l.,

(2 01

7a , (

b) ; M

ar in

o et

 a l.,

(2 02

1) ; P

an dy

a, (2

02 1)

; S ch

w ar

tz m

an e

t a l.,

(2

02 2)

; V al

er o

et  a

l., (2

02 2)

Ps yc

ho lo

gi ca

l fl ex

ib ili

ty (n

= 6)

A cc

ep ta

nc e

an d

A ct

io n

Q ue

sti on

na ire

— Se

co nd

v er

si on

(A A

Q -I

I; B

on d

&

B un

ce , (

20 03

)) (n

= 5)

Ç iç

ek G

üm üş

& Ö

nc el

, ( 20

22 );

H ah

s e t a

l., (2

01 9)

; M ar

in o

et  a

l., (2

02 1)

; Sc

hw ar

tz m

an e

t a l.,

(2 02

2) ; W

hi tti

ng ha

m e

t a l.,

(2 02

2) Pa

re nt

al A

cc ep

ta nc

e Q

ue sti

on na

ire (6

-P A

Q ; G

re en

e et

 a l.,

(2 01

5) ) (

n = 1)

Lo ba

to e

t a l.,

(2 02

3) M

in df

ul p

ar en

tin g

(n =

4) Th

e In

te rp

er so

na l M

in df

ul ne

ss in

P ar

en tin

g (I

M P;

D un

ca n

et  a

l., (2

00 9)

) Lo

e t a

l., (2

01 7a

, b );

Si eb

el in

k et

 a l.,

(2 01

8) ; W

hi tti

ng ha

m e

t a l.,

(2 02

2) M

in df

ul aw

ar en

es s (

n = 4)

M in

df ul

A tte

nt io

n A

w ar

en es

s Sc

al e

(M A

A S;

B ro

w n

et  a

l., (2

00 3)

) ( n =

4) H

ah s e

t a l.,

(2 01

9) ; L

iu e

t a l.,

(2 02

1) ; M

ar in

o et

 a l.,

(2 02

1) ; S

ch w

ar tz

m an

e t a

l.,

(2 02

2)

3325Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

Ta bl

e 2

S um

m ar

y of

fi nd

in gs

*p <

0. 05

; * *p

< 0.

00 1

O ut

co m

es M

et a-

an al

ys es

N ar

ra tiv

e sy

nt he

se s

C er

ta in

ty o

f th

e ev

id en

ce N

o. o

f pa

rti ci

– pa

nt s

SM D

(9 5%

C I)

Re fe

re nc

es N

o. o

f pa

rti ci

– pa

nt s

H ed

ge s’

g Re

fe re

nc es

Pa re

nt al

st re

ss 10

15 −

 0 .6

9 (−

 1 .0

5, −

 0 .3

3) B

eh ba

ha ni

e t a

l., (2

01 8)

; C hr

on is

e t a

l., (2

00 6)

; F ei

nb er

g et

 a l.,

(2 01

4) ; F

er ra

io li

an d

H ar

ris , (

20 13

); H

o et

 a l.,

(2

02 1)

; L iu

e t a

l., (2

02 1)

; L o

et  a

l., (2

01 7a

, 2 01

7b );

M ar

in o

et  a

l., (2

02 1)

; O ny

is hi

e t a

l., (2

02 3)

; S ch

w ar

tz –

m an

e t a

l., (2

02 2)

; S ha

rif e

t a l.,

(2 01

5) ; V

al er

o et

 a l.,

(2

02 2)

; W hi

tti ng

ha m

e t a

l., (2

02 2)

; W on

g et

 a l.,

(2 01

0)

13 7

60 46

−  8

.6 4*

* −

 5 .8

1* *

−  0

.7 0*

Pa nd

ya , (

20 21

) Ç

iç ek

G üm

üş a

nd Ö

nc el

, ( 20

22 )

N ee

ce , (

20 14

)

M od

er at

e

D ep

re ss

iv e

sy m

pt om

s 92

5 −

 0 .9

5 (−

 1 .4

7, −

 0 .4

3) C

hr on

is e

t a l.,

(2 00

6) ; F

ei nb

er g

et  a

l., (2

01 4)

; H ah

s e t a

l.,

(2 01

9) ; K

ho sh

va gh

t e t a

l., (2

02 1)

; K uh

lth au

e t a

l.,

(2 02

0) ; L

iu e

t a l.,

(2 02

1) ; L

o et

 a l.,

(2 01

7a , 2

01 7b

); O

ny is

hi e

t a l.,

(2 02

3) ; S

ch w

ar tz

m an

e t a

l., (2

02 2)

; Sh

ar eh

a nd

Y az

da ni

an , (

20 23

); Sh

ar if

et  a

l., (2

01 5)

; W

hi tti

ng ha

m e

t a l.,

(2 02

2)

24 3

46 60

−  1

.0 5*

* −

 0 .8

7* −

 5 .0

4* *

D yk

en s e

t a l.,

(2 01

4) N

ee ce

, ( 20

14 )

Ç iç

ek G

üm üş

a nd

Ö nc

el , (

20 22

)

H ig

h

A nx

ie ty

48 4

−  0

.7 8

(−  1

.3 9,

−  0

.1 8)

C hr

on is

e t a

l., (2

00 6)

; K ho

sh va

gh t e

t a l.,

(2 02

1) ;

K uh

lth au

e t a

l., (2

02 0)

; L iu

e t a

l., (2

02 1)

; O ny

is hi

et

 a l.,

(2 02

3) ; S

ch w

ar tz

m an

e t a

l., (2

02 2)

; S ha

rif e

t a l.,

(2

01 5)

; W hi

tti ng

ha m

e t a

l., (2

02 2)

24 3

60 −

 0 .9

0* *

−  2

.7 1*

* D

yk en

s e t a

l., (2

01 4)

Ç iç

ek G

üm üş

a nd

Ö nc

el , (

20 22

) M

od er

at e

Pa re

nt al

d ist

re ss

83 8

−  0

.2 9

(−  0

.4 2,

−  0

.1 5)

B eh

ba ha

ni e

t a l.,

(2 01

8) ; H

o et

 a l.,

(2 02

1) ; K

uh lth

au

et  a

l., (2

02 0)

; L iu

e t a

l., (2

02 1)

; L o

et  a

l., (2

01 7a

, 20

17 b)

; M ar

in o

et  a

l., (2

02 1)

; S ch

w ar

tz m

an e

t a l.,

(2

02 2)

; S ha

re h

an d

Ya zd

an ia

n, (2

02 3)

; S ie

be lin

k et

 a l.,

(2

01 8)

; V al

er o

et  a

l., (2

02 2)

13 7

46 60

−  6

.7 **

−  0

.7 0*

−  5

.5 0*

*

Pa nd

ya , (

20 21

) N

ee ce

, ( 20

14 )

Ç iç

ek G

üm üş

a nd

Ö nc

el , (

20 22

)

H ig

h

Pa re

nt al

w el

l-b ei

ng 42

6 0.

62 (0

.2 0,

1 .0

3) (F

er ra

io li

an d

H ar

ris , (

20 13

); H

o et

 a l.,

(2 02

1) ; L

o et

 a l.,

(2

01 7a

, 2 01

7b );

Lo ba

to e

t a l.,

(2 02

3) ; S

ha rif

e t a

l.,

(2 01

5) ; S

ie be

lin k

et  a

l., (2

01 8)

; W hi

tti ng

ha m

e t a

l.,

(2 02

2) ; W

on g

et  a

l., (2

01 0)

– –

– M

od er

at e

Pa re

nt –c

hi ld

re la

tio ns

hi p

61 5

0. 43

(0 .2

2, 0

.6 4)

B eh

ba ha

ni e

t a l.,

(2 01

8) ; C

hr on

is e

t a l.,

(2 00

6) ; H

o et

 a l.,

(2

02 1)

; L iu

e t a

l., (2

02 1)

; L o

et  a

l., (2

01 7a

, 2 01

7b );

Lo

et  a

l., (2

01 7a

, 2 01

7b );

M ar

in o

et  a

l., (2

02 1)

; S ch

w ar

tz –

m an

e t a

l., (2

02 2)

; V al

er o

et  a

l., (2

02 2)

13 7

5. 49

** Pa

nd ya

, ( 20

21 )

H ig

h

M in

df ul

p ar

en tin

g 43

4 0.

15 (−

 0 .0

5, 0

.3 5)

Lo e

t a l.,

(2 01

7a , 2

01 7b

); Si

eb el

in k

et  a

l., (2

01 8)

; W hi

t- tin

gh am

e t a

l., (2

02 2)

– –

– M

od er

at e

M in

df ul

aw ar

en es

s 18

5 1.

99 (−

 0 .6

4, 4

.6 2)

H ah

s e t a

l., (2

01 9)

; L iu

e t a

l., (2

02 1)

; M ar

in o

et  a

l.,

(2 02

1) ; S

ch w

ar tz

m an

e t a

l., (2

02 2)

– –

– Ve

ry lo

w

Ps yc

ho lo

gi ca

l fl ex

ib ili

ty 13

8 1.

47 (−

 0 .4

2, 3

.3 6)

H ah

s e t a

l., (2

01 9)

; L ob

at o

et  a

l., (2

02 3)

; M ar

in o

et  a

l.,

(2 02

1) ; S

ch w

ar tz

m an

e t a

l., (2

02 2)

; W hi

tti ng

ha m

e t a

l.,

(2 02

2)

60 6.

08 **

Ç iç

ek G

üm üş

a nd

Ö nc

el , (

20 22

) Ve

ry lo

w

3326 Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

Fi g.

3

Fo re

st pl

ot s e

ffe ct

o f C

B Is

o n a

pa re

nt al

st re

ss ; b

d ep

re ss

iv e

sy m

pt om

s o f p

ar en

ts ; c

a nx

ie ty

o f p

ar en

ts ; d

p ar

en ta

l d ist

re ss

3327Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

Fi g.

4

Fo re

st pl

ot s e

ffe ct

o f C

B Is

o n a

su bj

ec tiv

e w

el l-b

ei ng

; b p

ar en

t– ch

ild re

la tio

ns hi

p; c

m in

df ul

p ar

en tin

g; d

m in

df ul

aw ar

en es

s; e

p sy

ch ol

og ic

al fl

ex ib

ili ty

3328 Journal of Autism and Developmental Disorders (2024) 54:3316–3335

1 3

analysis confirmed the robustness of the result (Supple- mentary Fig. 8).

Parent‒Child Relationship The parent‒child relationship was examined by nine studies with 615 participants (Behba- hani et al., 2018; Chronis et al., 2006; Ho et al., 2021; Liu et al., 2021; Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b; Marino et al., 2021; Schwartzman et al., 2022; Valero et al., 2022). CBIs had a small effect on the parent‒child rela- tionship (g = 0.43, 95% CI [0.22, 0.64], P = 0.15, I2 = 33%; Fig. 4b). Furthermore, the result was not altered after per- forming a sensitivity analysis (Supplementary Fig. 9).

Mindful Parenting Four trials involving 434 participants reported the effects of CBIs on mindful parenting (Lo et al., 2017a, 2017b; Lo et al., 2017a, 2017b; Siebelink et al., 2018; Whittingham et al., 2022). The findings demonstrated that CBIs did not substantially promote mindful parenting (g = 0.15, 95% CI [− 0.05, 0.35], P = 0.40, I2 = 0%; Fig. 4c). The sensitivity analysis confirmed that these findings were reliable (Supplementary Fig. 10).

Mindful Awareness Four trials with 185 subjects were used to investigate the influence of CBIs on mindful aware- ness (Hahs et al., 2019; Liu et al., 2021; Marino et al., 2021; Schwartzman et al., 2022). No significant improve- ments were revealed in the pooled results (g = 1.99, 95% CI [− 0.64, 4.62], P < 0.01, I2 = 88%; Fig. 4d); however, the results of the sensitivity analysis revealed the potential benefit of CBIs after removing an outlier (g = 0.61, 95% CI [0.27, 0.96], P < 0.01, I2 = 0%; Supplementary Fig. 11) (Marino et al., 2021).

Psychological Flexibility A total of five studies includ- ing 138 participants were used to examine the effect of CBIs on psychological flexibility (Hahs et al., 2019; Lobato et al., 2023; Marino et al., 2021; Schwartzman et al., 2022; Whittingham et al., 2022). The results of the meta-analysis revealed that the CBIs that were implemented in those stud- ies did not significantly enhance psychological flexibility (g = 1.47, 95% CI [− 0.42, 3.36], P < 0.01, I2 = 85%; Fig. 4e). However, the favourable effect of CBIs was discovered after excluding one outlier (g = 0.43, 95% CI [0.03, 0.83], P = 0.14, I2 = 0%; Supplementary Fig. 12) (Marino et al., 2021).

Comparison of Treatment Effects Between Different Intervention Approaches

This subgroup analysis did not include mindful aware- ness, or psychological flexibility due to the paucity of relevant research. The subgroup analysis indicated that MBIs had a significant effect on parental stress levels (knumber of outcomes = 8, g = − 0.50, 95% CI [− 0.78, − 0.22], P = 0.03, I2 = 55%), depressive symptoms (k = 3, g = − 0.38, 95% CI [− 0.60, − 0.17], P = 0.72, I2 = 0%), parental distress levels (k = 8, g = − 0.24, 95% CI [− 0.39, − 0.08], P = 0.59,

I2 = 0%), parental well-being levels (k = 4, g = 0.42, 95% CI [0.17, 0.68], P = 0.15, I2 = 44%), and parent‒child rela- tionships (k = 6, g = 0.37, 95% CI [0.16, 0.58], P = 0.18, I2 = 34%). And CBT could also effectively decrease these parents’ parental stress levels (k = 5, g = − 1.02, 95% CI [− 1.89, − 0.15], P < 0.01, I2 = 93%) and depressive symp- toms (k = 5, g = − 1.31, 95% CI [− 2.28, − 0.34], P < 0.01, I2 = 94%), while improving parental well-being (k = 2, g = 1.20, 95% CI [0.31, 2.09], P = 0.03, I2 = 79%). The results of subgroup analysis for different interventions are displayed in Figs.3 and 4.

Comparison of Treatment Effects Between Parent‑Only and Parent‒Child Dyad CBIs

A subgroup analysis revealed that parent‒child dyad CBIs were more favourable than parent-only CBIs in improving parent‒child relationships (g = 0.66, 95% CI [0.15, 1.16] vs. 0.33, 95% CI [0.15, 0.51]; Supplementary Fig. 15), while parent-only CBIs were more beneficial than parent‒child dyad CBIs in decreasing parental stress levels (g = − 0.73, 95% CI [− 1.16, − 0.30] vs. − 0.57 [− 1.18, 0.04]; Supple- mentary Fig. 13), distress levels (g = − 0.29, 95% CI [− 0.51, − 0.10] vs. − 0.28, 95% CI [− 0.55, − 0.00]; Supplementary Fig. 14), and enhancing parental well-being (g = 0.76, 95% CI [0.24, 1.29] vs. 0.30, 95% CI [− 0.19, 1.03]; Supplemen- tary Fig. 16).

Comparison of Treatment Effects Between Intervention Durations

According to the subgroup analyses, the ideal intervention duration was 1 to 8 weeks for decreases in parental stress levels (k = 7, g = − 0.60, 95% CI [− 0.95, − 0.25], P = 0.03, I2 = 58%; Supplementary Fig. 17), depressive symptoms (k = 5, g = −  0.91, 95% CI [−  1.63, −  0.18], P < 0.01, I2 = 82%; Supplementary Fig. 18), anxiety levels (k = 3, g = − 0.97, 95% CI [− 1.43, − 0.51], P = 0.25, I2 = 27%; Supplementary Fig. 19), and parental distress levels (k = 7, g = − 0.30, 95% CI [− 0.49, − 0.11], P = 0.25, I2 = 23%; Sup- plementary Fig. 20), as well as for improvements in paren- tal well-being levels (k = 4, g = 0.49, 95% CI [0.22, 0.75], P = 0.33, I2 = 13%; Supplementary Fig. 21), parent‒child relationships (k = 5, g = 0.59, 95% CI [0.21, 0.97], P = 0.03, I2 = 62%; Supplementary Fig. 22), mindful awareness (k = 2, g = 0.88, 95% CI [0.31, 1.45], P = 0.52, I2 = 0%; Supplemen- tary Fig. 23), and parental psychological flexibility (k = 3, g = 0.69, 95% CI [0.18, 1.19], P = 0.98, I2 = 0%; Supplemen- tary Fig. 24). However, significant effects on parental stress levels (k = 8, g = − 0.43, 95% CI [− 0.72, − 0.15], P = 0.03, I2 = 54%; Supplementary Fig. 17) and parent‒child relation- ships (k = 4, g = 0.28, 95% CI [0.01, 0.55], P = 0.93, I2 = 0%;

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Supplementary Fig. 22) were also observed when the inter- vention duration was over eight weeks.

Comparison of Treatment Effects Between Two Types of DD in Children

No subgroup analyses were conducted for children with CP, ID, or blended types of DD due to the limited num- ber of relevant studies. The subgroup analysis found that CBIs had significantly positive effects on parental stress levels (k = 7, g = − 0.60, 95% CI [− 0.84, − 0.36], P = 0.18, I2 = 32%; Supplementary Fig. 25), parental distress lev- els (k = 5, g = − 0.33, 95% CI [− 0.52, − 0.13], P = 0.66, I2 = 0%; Supplementary Fig. 28), parental well-being levels (k = 4, g = 0.79, 95% CI [0.25, 1.33], P < 0.01, I2 = 77%; Sup- plementary Fig. 29), and parent‒child relationships (k = 5, g = 0.43, 95% CI [0.14, 0.73], P = 0.14, I2 = 43%; Supple- mentary Fig. 30) for parents of children with ADHD. In addition, CBIs were shown to effectively reduce parental stress levels (k = 7, g = − 0.99, 95% CI [− 1.79, − 0.20], P < 0.01, I2 = 91%; Supplementary Fig.  25), depressive symptoms (k = 5, g = − 0.99, 95% CI [− 1.83, − 0.15], P < 0.01, I2 = 91%; Supplementary Fig. 26), and anxiety levels (k = 3, g = − 1.40, 95% CI [− 2.62, − 0.17], P < 0.01, I2 = 92%; Supplementary Fig. 27) for parents of children with ASD.

Narrative Syntheses

Four studies (Dykens et al., 2014; Pandya, 2021) (Çiçek Gümüş & Öncel, 2022; Neece, 2014) conducted narrative analyses. When compared to the positive psychotherapy group, Dykens et al. (2014) observed substantial postint- ervention reductions in depression (g = 1.05) and anxiety (g = 0.90) symptoms for parents of children with DD using an in-person MBI. Moreover, Pandya (2021) reported that utilizing online MBI versus a waitlist control group resulted in a considerable reduction in parental stress (g = − 8.64) distress (g = − 6.70) levels, and a postintervention improve- ment in parent‒child relationships (g = 5.63). Furthermore, Neece (2014) identified that mindfulness-based stress reduc- tion could significantly alleviate parental distress (Cohens’ d = 0.70) and depression (d = 0.87) for parents of children with DD postintervention compared to the wait-list con- trol group. In addition, Çiçek Gümüş and Öncel (2022) found significant reductions in parental stress (g = − 5.81), depressive symptoms (g = − 5.04), anxiety (g = − 2.71), parental distress (g = − 5.50), and psychological flexibility (g = − 6.08) for parents of children with ASD and mental disorder(s) postintervention when comparing ACT to the TAU control group.

Risk of Bias

In general, one study was determined to have a high risk of bias (Behbahani et al., 2018), while the other studies were judged to have some concerning aspects regarding the risk of bias (Figure 2). Due to a lack of information on allo- cation concealment, 13 studies (52%) were found to have some concerning aspects regarding a risk of bias resulting from the randomization technique, whereas the other studies showed low risk. A total of 18 studies (72%) were revealed to have some concerning aspects regarding deviations from the intended intervention, while the remaining seven stud- ies were deemed to be low risk. Except for one study, which was deemed high risk due to no information concerning insufficient/missing data, other studies were deemed low risk (96%). All studies were noted to have some concern- ing aspects regarding outcome measurements because they all used self-reported instruments. In the selection of the published results, 13 studies (52%) were rated as low risk, while the remaining studies were evaluated as having some concerning aspects due to inadequate information regarding prespecified analytical plans/protocols.

Quality of the Evidence

The aggregate GRADE evaluation of the outcomes revealed that the certainty of evidence results ranged from “high” to “very low” (Table 1). The effects of CBIs on parental distress, depression levels, and parent‒child relationships were determined to have a high certainty of evidence. The evidence regarding effects on parental stress and anxiety lev- els, parental well-being, and mindful parenting was judged as having moderate certainty of evidence because of signifi- cant heterogeneity or inconsistency. The evidence regarding effects on mindful awareness and psychological flexibility had a low level of certainty, mainly because of the risk of bias, inconsistency (high heterogeneity), and/or imprecision (sample size lower than 400) of the relevant studies.

Discussion

This review and meta-analysis summarized and synthesized 25 RCTs to examine the effectiveness of CBIs in reducing distress levels and improving mental health and wellbe- ing among parents of children with DD. The results of the meta-analysis highlight that CBIs may significantly allevi- ate the levels of parental stress, depressive symptoms, anxi- ety, and parental distress and improve parental well-being and parent‒child relationships for parents of children with DD. Moreover, MBIs and CBT interventions showed posi- tive effects and were recommended as potential optimal approaches in the subgroup analyses. The findings of our

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review echo those of previous systematic reviews and meta- analyses that CBIs had positive effects for reducing parental stress and psychological symptoms postintervention in par- ents of children with DD. Osborn et al., (2021) explored the impact of mindfulness therapies on psychological distress in parents of children with DD and reported small to large ESs (g range: 0.39 to 1.94). Lee et al. (2022) examined the effect of mindfulness parent training on parenting stress levels and found small to large ESs (g range: 0.06 to 0.84) for parents of children with ADHD. Hartley et al. (2019) conducted a meta-analysis on MBIs for parents of children with ASD and revealed postintervention improvement in subjective well- being. Bourke-Taylor et al. (2021) performed a meta-analysis on group interventions for mothers of children with disabili- ties and found that CBT demonstrated significantly large postintervention ESs for parenting stress levels (g = 0.86) and mental health (g = 1.14). As MBIs and CBT interven- tions have been identified as optimal approaches, future well-designed RCTs are needed to confirm their effects on parents of children with DD and, equally importantly, the factors and mechanisms of action impacting their efficacy.

However, this review did not identify significant improve- ment in the outcomes of mindfulness parenting, mindfulness awareness, and psychological flexibility postintervention for parents of children with DD, which is in line with Chua and Shorey’s (2022) meta-analysis, which examined the effec- tiveness of MBIs and ACT interventions among parents of children with DD. The findings of the current study sug- gest that cautious interpretation is needed due to the limited number of studies and the small sample sizes included in the meta-analysis, as well as the heterogeneity of the interven- tion content. Another reason for the resulting ineffective- ness is that those studies did not set mindfulness parenting, mindfulness awareness, and/or psychological flexibility as primary outcomes. The intervention may only indirectly impact those outcomes, implying that some outcomes need to be sufficiently addressed with current CBIs. In contrast, Rayan and Ahmad (2018) found that five out of six trials reported postintervention improvements in mindful parent- ing among parents of children with disabilities, although only a narrative synthesis was performed in that study and only one RCT was included, implying that direct compari- sons could not be conducted. Similarly, studies included in our study indicated that CBIs could facilitate mindful aware- ness and psychological flexibility outcomes, but the opposite pooled result was obtained after performing meta-analyses. This may be because we applied the random-effects model to account for the significant heterogeneity since one study (Marino et al., 2021) displayed an extremely large ES for these two outcomes. The positive effect appeared for mindful awareness (g = 0.70, 95% CI [0.39, 1.01], P < 0.01, I2 = 88%) and psychological flexibility (g = 0.56, 95% CI [0.19, 0.92], P < 0.01, I2 = 85%) after we switched to the fixed-effects

model. Therefore, additional studies are required to further explore the potential benefits of CBIs on these inconsistent outcomes.

Moreover, Chua and Shorey’s (2022) meta-analysis also demonstrated that MBIs and ACTs were effective in reduc- ing parental stress, anxiety, and depression, which supported the findings of our study discussed above; however, our pooled results showed that no significant reductions were observed for parental stress or depressive symptoms when using ACT for parents of children with DD. This may be due to the paucity of research investigating the effectiveness of ACT for parents of children with DD, which allowed for only 4 eligible studies to be included in our meta-analysis. All included studies concluded that ACT could be applied to lessen depressive symptoms, anxiety, and parental dis- tress and to promote parental well-being, parent‒child rela- tionships, mindful parenting, and mindful awareness, even though the conclusions for parental stress and psychological flexibility were inconsistent. The reason that these two out- comes did not change in the RCT conducted by Whittingham et al. (2022) may be because some of the parents of children with CP had to cope with not only their children’s motor problems but also other problems caused by comorbidi- ties, such as epilepsy (14.93%), ASD (8.96%), and ADHD (4.48%), resulting in a higher level of stress and making it more difficult to actively develop personal growth (e.g., psychological flexibility) (Li et al., 2016). Furthermore, as Chua and Shorey (2022) did not further conduct a subgroup analysis to explore the effectiveness of MBIs and ACT sepa- rately, a direct comparison could not be drawn. In addition, we did not perform a subgroup analysis for DBT and CFT because only one relevant study was included in our review, respectively. However, Shareh et al. (2023) revealed that DBT significantly reduced distress and depression in moth- ers of children with ID postintervention compared with the wait-list control. And Khoshvaght et al. (2021) demonstrated that CFT effectively alleviated anxiety and depression in mothers of children with CP postintervention compared with no treatment. As a result, future well-designed RCTs are recommended to examine and confirm the effectiveness of ACT, DBT, and CFT on the outcomes mentioned above for parents of children with DD as well as to investigate the broad potential to confer meaningful benefits to these par- ents beyond those outcomes.

Apart from the impact of the intervention approach men- tioned above, other moderators, including intervention dura- tion, targeted participants, and the types of DD among the children, also revealed a significant impact on the outcomes. Consistent with previous studies showing that eight weeks of MBIs (Gotink et al., 2016) and no more than five ACT sessions (Li et al., 2021) can significantly change various health indicators postintervention, our review found that 1 to 8 weeks was regarded as the optimal duration of CBIs

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for parents of children with DD to reduce parental anxiety and improve parental well-being, parent‒child relationships, mindful awareness, psychology flexibility. Furthermore, for participant targets, including parent‒child dyads and par- ent-only participants, parent-only participants showed more positive effects for the CBI outcomes (e.g., parental stress, distress, and well-being) than parent‒child dyads. However, parent–child dyads showed greater ES than parent-only par- ticipants in parent–child relationships. This may be because parent‒child dyads could improve parent‒child interactions, thus further alleviating relational frustration, parental stress, and other mental health problems (Dennis et al., 2018). Moreover, subgroup analyses also demonstrated that CBIs were associated with significant improvements for parents of children with two different types of DD (ADHD and/or ASD) in parental stress and other outcomes, which is in line with previous studies (Hartley et al., 2019; Lee et al., 2022; Yu et al., 2019). However, considering the unique needs and experiences among parents of children with different disease types, future studies and research should be syndrome- and disease-specific. Furthermore, these subgroup analyses were complicated by the fact that format was confounded with different intervention approaches. Therefore, explore other variable that potentially affect the outcomes is another rec- ommendation for future research to consider.

There are several limitations to consider. First, the gender imbalance among the participants, which may limit findings to mother-specific experiences, should be taken into account. The proportion of women in the sample largely reflects that mothers have historically been the primary caregivers of children with DD. However, given that there is evidence to suggest that parenting experiences, mental health, and stress outcomes may differ for fathers of children with DD (Seymour et al., 2018), future studies should consider these gender differences.

Second, because many potential moderators were not dis- closed and/or reported inconsistently across studies, such as sociodemographic factors (e.g., child and parent ages, gender, race/ethnicity, family income, child symptom sever- ity and functioning), intervention characteristics (e.g., inter- vention context, dosage, number of sessions, and delivery location), and measurement tools, this review was unable to evaluate them all. These moderators/variations may bring further significant heterogeneity into the meta-analyses. Therefore, more consistent reporting of these variables is required for future studies to meaningfully examine system- atic moderators.

Third, a total of eight studies (38.10%) were pilot studies with a small sample size (of which the number of participants in six studies was ≤ 20 per group), which may not provide enough statistical power to identify intervention effects and thus may hinder the internal and

external validity of study outcomes. Moreover, no study was assessed as having a low risk of bias, and the majority of included studies had some methodological limitations, such as a lack of information about allocation conceal- ment, intention-to-treat analyses, and prespecified proto- cols, as well as unblinding the intervention to participants and intervenors and the use of self-report questionnaires. In addition, the effects of CBIs on some of the apparently unaffected outcomes, such as mindful parenting, mindful awareness, and psychological flexibility, still need to be determined due to the paucity of relevant studies. There- fore, future research with rigorously designed RCTs is recommended with different intervention approaches and with the objective of extending the list of outcomes to include currently unclear outcomes to support parents of children with DD.

Finally, this study did not conduct subgroup analysis for comparison between parents of children with medical comorbidity (such as ASD-ID, ADHD-ID, ASD-ADHD, ASD-epilepsy) versus ASD as a single medical condition, or between parents of children with or without ID. This restriction was due to lack of such information reported in the included studies. This restriction is likely to limit the generalizability of the findings reported here across all those diagnosed with ASD. One factor relevant to this has been the changes in this population resulting from changes to diagnostic criteria. More specifically, researchers using data from parents whose children were diagnosed prior to 2017 and who were funded by the National Institute of Mental Health (NIMH) in the United States were required to ensure that their sample met DSM-IV or IV-Tr criteria. These criteria explicitly excluded children from diagno- sis with ASD-ADHD comorbidity; this restriction was removed in 2017 with the release of DSM-5, which per- mitted comorbidity and this subgroup now constitutes a significant part of the ASD population. In addition, Asper- ger syndrome which was previously coded separately, became part of the general ASD category. Asperger syn- drome constituted a significant group of children without significant developmental delay or intellectual difficulties, and a reduced level of medical comorbidity. These broad- ening of criteria mean that post- 2017 parents were likely to experience more diverse levels of distress and caregiver burden. For families with DD children, more healthcare, education, and social support services, are often required and it is essential to identify them, to allocate additional resource. We also need to design and evaluate syndrome- and disease-specific interventions. It is recommended, therefore that a proportion of future studies are focussed on these specific populations, clearly describe the eligibil- ity criteria of their participants, and use this framework to develop interventions that best meet the specific needs of their parents’ in caring for themselves and their children.

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Conclusion

The positive effects of CBIs on parental stress levels, depres- sive symptoms, anxiety levels, parental distress, parental well-being, and parent–child relationships were highlighted in this review. Factors that influenced intervention effects included intervention approaches and durations, participant targets, and the types of DD among the children. The current evidence should be strengthened by additional well-designed RCTs that explore and examine the process, predictive fac- tors, and mechanism of action of the best interventions for parents of children with DD.

Supplementary Information The online version contains supplemen- tary material available at https:// doi. org/ 10. 1007/ s10803- 023- 06063-x.

Acknowledgments None.

Author Contributions Sini Li contributed to records identification, data extraction and curation, formal analysis, and writing and revising manuscripts. Yijing Yong contributed to records identification, data extraction and curation. Yamin Li contributed to records identifica- tion, data extraction and curation, and obtaining funding. Jianhe Li and Jiao Xie contributed to Conceptualisation, methodology, supervision, visualization, final review and editing.

Funding The research was funded by the National Natural Science Foundation of China (No: 81873806).

Data Availability All data generated or analysed during this study are included in this published article as Supplementary information files.

Declarations

Conflict of interest There is no conflict of interest between all authors.

Ethical Approval and Consent to Participate This is a systematic review and meta-analyses for which the patient data were all obtained from previous published studies. It does not contain any studies with animal or human participants.

Open Access This article is licensed under a Creative Commons Attri- bution 4.0 International License, which permits use, sharing, adapta- tion, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/.

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Brief Report

Potential for children with intellectual disability to engage in cognitive behaviour therapy: the parent perspective

A. Hronis,1 R. Roberts,2 L. Roberts1 & I. Kneebone1

1 Graduate School of Health, University of Technology Sydney, Ultimo, Australia 2 The University of Adelaide, Adelaide, Australia

Abstract

Background This study aimed to obtain the opinions of parents and carers of children with intellectual disability (ID) as to whether cognitive behaviour therapy (CBT) could be useful for their children. Methods A mixed qualitative and quantitative method was employed. Twenty-one carers of children aged 10 to 17 having borderline to moderate intellectual functioning responded to an online questionnaire. Participants were provided with information about CBT and asked to respond to open-ended questions. Quantitative data pertained to questions about their child’s ability to identify and describe thoughts, feelings and behaviours. Thematic analysis of responses was conducted using an inductive method of identifying themes from the qualitative data collected. Results Five themes emerged from the qualitative analysis: Emotional Attunement (i.e. parent’s understanding and recognition of their child’s emotions), Role of the Therapist (i.e. ways therapists could facilitate the intervention), Role of the Parent (i.e. ways parents could engage in the therapy process), Anticipated Obstacles (i.e. what may get in the way of the therapy) and Suggested Adaptations for

Therapy (i.e. how CBT can be adapted to suit the needs of children with ID). Seventy-six per cent agreed that their child would be able to engage in CBT with assistance. Conclusions The majority of parents believed that CBT is an intervention that children with ID could engage in, provided the therapy is adapted, and the therapist accommodates their needs.

Keywords children, cognitive behaviour therapy, intellectual disability, learning disability

Background

Up to 50% of children with intellectual disability (ID) have a comorbid mental illness (Einfeld et al. 2011; Tonge & Einfeld 2000). Because of deficits in intellectual functioning, treatments have largely involved behavioural interventions and use of medications (Vereenooghe & Langdon 2013). Recently, cognitive behaviour therapy (CBT) has been identified as an effective treatment for adults with mild to moderate ID and comorbid depression, anxiety and anger (e.g. Hassiotis et al. 2013; Osugo & Cooper 2016; Roberts & Kwan 2018; Vereenooghe & Langdon 2013). While similar trials have not been conducted among children with ID, CBT could be a treatment option for children with ID, provided adaptations are made (Hronis et al. 2017).

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Correspondence: Ms Anastasia Hronis, Discipline of Clinical

Psychology, Graduate School of Health, University of Technology

Sydney, PO BOX 123, Broadway NSW 2007, AUSTRALIA. (e-mail:

[email protected])

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Neuropsychological deficits for children with ID have been identified in learning, memory, attention, executive functions and language. The impact on therapy and adaptations for therapy has been proposed specific to CBT (see Hronis et al. 2017). Cognitive behaviour therapy is the gold standard

intervention for many psychopathologies for children without ID (e.g. Cartwright-Hatton et al. 2004; Compton et al. 2004). At the foundation of CBT is the identification and linking of thoughts, emotions and behaviours, which adults with ID can do (Dagnan et al. 2000; Joyce et al. 2006; Oathamshaw & Haddock 2006; Sams et al. 2006). To our knowledge, this has not been evaluated among children with mild to moderate ID. Parents play a crucial role in the therapy process for children with and without developmental disabilities (Manassis et al. 2014; White et al. 2010). Involving parents is in line with family-centred practices for interventions for children, recognising that parent-therapist collaboration in planning and evaluating interventions is key, and based on the principle that parents know their child best (Hanna & Rodgers, 2002; Rosenbaum et al. 1998). The aim of this study was to gather the opinions of

parents who have children with ID about CBT. The researchers set out to understand if parents who have children with ID believe their child could engage in the process of CBT and factors associated with this. As to our knowledge, no research has previously investigated this, the study was exploratory.

Method

Procedure

This research was approved by the University Ethics Committee (approval number: 2015000482–54). Participants were recruited via online advertising through mental health organisations and social media. The study was advertised as seeking parents/carers of children with a mild to moderate ID, aged 10 to 17, to respond to questions online about how their child thinks and feels.

Measures

Parent report of child’s ability to engage in CBT

Participants read information about CBT and rated their child’s ability to express feelings, articulate

thoughts, describe actions and link thoughts, feelings and behaviours. Parents rated on a 5-point Likert scale how often they know if their child is feeling happy, sad, angry and anxious/worried.

Emotions development questionnaire – parent form (Wong et al. 2009)

The Emotions Development Questionnaire – Parent Form (EDQ-P) assesses emotional understanding, emotional and behavioural regulation, theory of mind and problem solving in children with autism spectrum disorder, with or without ID (Ratcliffe et al. 2014). Quantitative data were used to supplement qualitative data to describe the emotional development of the sample. It has 29 items rated on a 5-point Likert scale, added to produce a total score. The EDQ-P has excellent internal consistency (α = .91; Ratcliffe et al. 2014), and was high in the current sample (α = 0.92). Higher scores indicate greater emotional understanding.

Open-ended questions

Participants were provided with information about the components of CBT, illustrated by a case example (Data S1), and responded to open-ended questions about their child’s potential to engage in CBT (Data S2).

Data analysis

Descriptive statistics for quantitative data were calculated. Thematic analysis was used to analyze the qualitative data. This involved initial prolonged engagement with the data via repeated readings, coding of responses by two independent researchers and codes then collated into themes (Braun & Clarke, 2006).

Results

Participants

Participants were 21 parents/carers of children in Australia between the ages of 10 and 17 with a mild or moderate ID or borderline intellectual functioning. The average age was 13.33 (SD = 2.58). Based upon parent reports, 23% had mild ID, 33% had moderate ID, 10% were on the border of mild to moderate ID, 5% had borderline intellectual functioning and 29%

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were unspecified. Comorbid diagnoses are reported in Table 1.

Quantitative data

Most participants reported they could often or almost always tell when their child was sad (76%), angry (76%) and happy (72%). 48% could often or almost always tell when their child was anxious or worried. One third agreed that their child can describe their

emotional state, while one third disagreed/strongly disagreed, and 24% were undecided (Figure 1). Fourteen per cent agreed their child can articulate their thoughts, while 43% disagreed/strongly disagreed, and one third were undecided. Twenty- four per cent agreed their child can describe their actions, however 33% disagreed/strongly disagreed, and one third were undecided. Finally, only 10% agreed that their child could link thoughts, feelings and behaviours, while 19% were undecided and over half (62%) disagreed/strongly disagreed. Overall, 76% agreed that their child would be able to participate in CBT with assistance. Seventeen parents completed the EDQ-P, with

scores ranging from 80 to 168 out of 200 (M = 108.1; SD = 19.9), indicating a range of emotional development within the sample.

Qualitative data

Length of responses ranged from a few words, to multiple sentences and were organised into five themes: (1) emotional attunement, (2) role of the

parent/carer, (3) role of the therapist, (4) anticipated obstacles and (5) suggested adaptations to therapy.

Emotional attunement

Responses reflected parents’ ability to recognise emotional states in their child. Indicators of a child’s emotions reported were body language, facial expressions, behaviours, verbal expressions of the emotion and increased or decreased social interactions.

Role of the parent/carer

Participants referenced taking on the role of the therapist outside of sessions, to help practice and generalise using strategies, as participants stated “I would become the teacher for the therapy and reaffirm therapy at home” and “help with homework”. Parents acknowledged they could provide practical support by “sit[ting] in on sessions” and “taking notes”. Emotional support they could provide involved encouragement to participate.

Role of the therapist

Therapist’s knowledge and experience working with children with ID were identified as important. One participant wrote, it would help “if the therapist was understanding of the disabilities my son has and had experience working with them”. Parents wanted therapists to understand the strengths and weaknesses of their child and adapt therapy, as one parent stated “the approach taken to engage a child needs to be carefully thought out prior to engagement to reduce the likelihood of shutdown during therapy”.

Anticipated obstacles

The difficulty of identifying and expressing thoughts and emotions was identified. Parents questioned whether CBT may be too complex with “too many steps in the process”. Rigid thinking was a potential obstacle, as one participant stated their child is “a literal person so won’t generalise”. Practical obstacles included time constraints, cost of therapy, geographical restrictions and the difficulty of “finding a clinician willing to work with intellectual disability”.

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Table 1 Rate of comorbid diagnoses in the sample

Diagnosis n %

Autism spectrum disorder 14 67 Attention deficit hyperactivity disorder 12 57 Specific learning disorder 7 33 Anxiety 6 29 Oppositional defiant disorder 3 14 Depression 2 10 Auditory processing disorder 1 5 Cerebral palsy 1 5 Epilepsy 1 5 Robertsonian translocation 1 5 Sensory processing disorder 1 5

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Suggested adaptations to therapy

Parents suggested how CBT could be adapted for their child’s needs (Table 2).

Discussion

The involvement of stakeholders is in line with patient-centred care best practices (Epstein & Street,

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Figure 1. Parents rating of child’s

ability for CBT skills. CBT,

cognitive behavioural therapy

[Colour figure can be viewed at

wileyonlinelibrary.com]

Table 2 Proposed adaptations to CBT by parents of children with ID

Adaptations Specific suggestions from parents

Providing instructions •Provide explicit instructions •Break instructions into small steps •Use stories to explain concepts and to provide examples •Use videos to explain and teach

“Teaching in baby steps” “Make it as simple as possible”

Prompts and cues •Use of visual cues •Colour coded charts as prompts and reminders

“Make a chart of feelings and thoughts so they can visualise them” “Behavioural cues to “lock in” lessons”

Check understanding •Confirm the child has understood before progressing to the next step “Making sure he understands how to do it before moving onto the next thing”

Practicing skills •Repeat each step multiple times •Repeat practices of skills •Practice with multiple examples “Getting him to show you” “Practice in therapy sessions and then practice outside of sessions”

Emotion training •Additional time to be spent on establishing an awareness and understanding of different emotional states “Teaching children to recognise feelings in the body that occur when getting close to a meltdown”

Support network •Have teachers involved in the treatment process •Have parents involved throughout treatment

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2011). To our knowledge, this is the first study exploring the carer perspective about how children with ID can engage in CBT. The results indicate that parents believe their child may benefit from CBT with assistance, provided therapy is adapted for their needs and barriers to treatment are managed. Although more than half of participants did not think their child could link thoughts, feelings and behaviours, three quarters agreed their child could participate in CBT with assistance. This is promising, because while parents acknowledge their child may not currently have those skills, they are hopeful their child could learn them. Parents provided suggestions on adapting CBT, which were consistent with those suggested by Hronis, Roberts and Kneebone (2017). Furthermore, parents were willing to take an active role in treatment, which is beneficial for CBT outcomes (Mendlowitz et al., 1999; Wood et al., 2009). A limitation was that parents who responded may

be more open to therapy, providing a possible biassed sample. Furthermore, the sample size was small, and some participants provided brief responses. Because of the nature of the online questionnaire, there was no opportunity to question further. Additionally, formal measures of intellectual and adaptive functioning were not used to confirm diagnoses. Nonetheless, the findings show promise for the use of CBT for children and adolescents with ID and mental health disorders. The results hold important practice implications and can contribute to the development and piloting of adapted CBT programmes for children with ID. Future research should focus on experimental studies exploring whether children with ID can make links between thoughts, feelings and behaviours and research trials evaluating the efficacy of adapted CBT for children with ID.

Conflict of Interest

The authors have declared that no conflict of interests exists.

Source of Funding

No external funding was received for the research reported in the paper.

Ethical Approval

This research was approved by the University Ethics Committee and undertaken with the understanding and written consent of each participant.

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Accepted 15 September 2019

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https://doi.org/10.1177/15407969221119848

Research and Practice for Persons with Severe Disabilities

2022, Vol. 47(3) 158 –175 © The Author(s) 2022

Article reuse guidelines: sagepub.com/journals-permissions

DOI: 10.1177/15407969221119848 rpsd.sagepub.com

Special Section Diversity, Equity, and Inclusion

Reconceptualizing Education Grounded in the Multimodal Discourses of Girls of Color Labeled with Significant Cognitive Disabilities

Amanda L. Miller1

Abstract The experiences of girls of color labeled with significant cognitive disabilities in middle school and high school have historically been excluded from educational research. This study sought to better understand how girls of color labeled with significant cognitive disabilities navigated multimodal discourses and classroom practices as well as how they were impacted by them. Using Disability Critical Race Theory and critical discourse theory, six students were focal participants and eight educators were secondary participants. Multiple case studies were used with primary (i.e., observations, audio/video recordings) and secondary (i.e., interviews, focus groups) data sources. Findings revealed how focal participants showed their discursive resourcefulness, despite absent communication supports and prioritization of oral/aural communication. Students also repositioned themselves in response to marginalization through talk and actions. Implications for research and practice are discussed. This study underscores the necessity of centering the experiences of girls of color labeled with significant cognitive disabilities in educational research to improve their school experiences.

Keywords girls of color labeled with significant cognitive disabilities, multimodal discourses, disability critical race theory (DisCrit), critical discourse theory, reimagining education

Multimodal discourses, including talk and actions, are inextricably connected to learning in schools. Youth engage in multimodal discourses throughout the day as they learn with and from peers and educa- tors (e.g., teachers, paraprofessionals). Talk may be verbalizations (e.g., “What do you think?”), vocal- izations (e.g., “Huh?,” “Oh,” “Eh”), or speech generated by a voice output device (Gee, 2014; Teachman et al., 2018). Actions or action-oriented expressions include eye gazes, facial expressions, gestures, and selections made on a communication board (Light & Drager, 2007; Scollon & Scollon, 2017). As such, students use talk and actions to explore concepts and content and to question and process (Rogoff, 2003).

Learning is a social process wherein students become active participants in knowledge communities (Lim & Renshaw, 2001). Youth labeled with significant cognitive disabilities have long been excluded from the social processes of learning. However, a mutually constitutive relationship exists between multimodal discourses and social practices for youth labeled with significant cognitive disabilities

1Wayne State University, Detroit MI, USA

Corresponding Author: Amanda L. Miller, Wayne State University, 285 College of Education, Detroit, MI 48202, USA. Email: [email protected]

1119848 RPSXXX10.1177/15407969221119848MillerResearch and Practice for Persons with Severe Disabilities research-article2022

Miller 159

(Wodak & Meyer, 2016). For example, when a student hears an educator continually praise a peer in class and condone another, then the student may attribute certain traits (e.g., smartness, goodness; Broderick & Leonardo, 2016) to one peer and not the other, based on how the educator responds. Moreover, these observations and interactions dictate how the student feels about their own responses and their willingness to take learning risks.

In addition, multimodal discourses mediate power relationships (Foucault, 1982). When students labeled with significant cognitive disabilities are not afforded opportunities to interact with peers, share knowledge, or construct meaning, then learning becomes a tool of inequity based on ideology. In response, youth may reposition or refuse to accept individual, group, and/or societal marginalization (Davies & Harre, 1990). They may speak out or act in another way when they notice educators and/or peers marginalizing one another (Annamma et al., 2020). Therefore, it is not only the educators and students who generate dis- courses, but the discourses also create who they are. In sum, a focus on multimodal discourses and social practices can illuminate how power and ideology are (re)produced in schools.

Context is crucial because of the constituted and constituting nature of thought, talk, and action. For example, the opportunities teachers create hold power over what exists and comes next in the classroom. When educators design opportunities for students to work with peers, then the knowledge the students explore and/or create together is influenced by each learner’s histories, perspectives, and experiences. That said, multimodal discourses can play a role in transforming society as students and educators use them in creative and agentic ways, thus exposing the dialectic nature of discourse and ideology (Gee, 2014). This can be true for the school experiences of girls of color labeled with significant cognitive disabilities; how- ever, these girls are often ignored in educational research (Sinclair et al., 2018) and their multimodal dis- courses are underrecognized and understudied. The term “girls of color” is used instead of “young women of color” to honor the experiences, expertise, and youthfulness of the girls in this project who identify as Afghan, Black, Hispanic, and Latina. While youth labeled with significant cognitive disabilities often expe- rience perpetual infantilization, the focal participants attended K–12 schools. Furthermore, childhood is often withheld from girls of color (Onyeka-Crawford et al., 2017).

While most of the multimodal discourse literature in education attends to educators (e.g., Berry, 2006a; Kurth et al., 2016; Orsati, 2014, 2015; Orsati & Causton-Theoharis, 2013), it is important to consider how students contribute to classroom discourses. For example, researchers have found student responding was unidirectional (i.e., students followed teacher directions, students answered teacher questions) rather than bidirectional (i.e., students asked questions, student–teacher, and student–student reciprocal conversations) in special education classrooms and segregated schools (Pennington & Courtade, 2015). Moreover, stu- dents with disabilities in special education classrooms have also experienced and engaged in disrespectful talk with their peers (Causton-Theoharis et al., 2011). Thus, students may unknowingly (re)produce harmful ideas about peer belonging, membership, and worth through their classroom discourses.

Scholars have also reported on how students with disabilities have repositioned or subtly claimed brief authority. For example, Black boys with disabilities repositioned by asking educators questions (e.g., “What did I do?”; Collins, 2011a) and sharing personal narratives (e.g., “I like to draw and paint.”; Collins, 2011b). Students with disabilities have also modified discussion topics or attempted to change an assumed turn- taking sequence with peers in the general education classroom (e.g., “Why are you taking over the paper?”; Berry, 2006b). In another study, one girl with autism persisted with personal narratives in a small group setting in response to her peers’ disinterest (e.g., “She talks to some Spanish people,”; Dean et al., 2013). Students with disabilities have also remained silent when urged to participate or have opted out (Collins, 2011b). Yet, more information is needed to understand how girls of color labeled with significant cognitive disabilities reposition multimodally in response to marginalization.

While little is known about the multimodal discourses of students labeled with significant cognitive dis- abilities, and girls of color specifically, the studies reviewed here provide a foundation. Few scholars have examined students’ multimodal discourses and many did not provide fine-grained details of their actions. Furthermore, student repositioning is a relatively newer point of inquiry, particularly for students labeled with significant cognitive disabilities. Therefore, additional research is needed to understand how intersecting oppressions at macrosociopolitical (e.g., ableism, racism; Erevelles & Minear, 2010) and microinteractional

160 Research and Practice for Persons with Severe Disabilities 47(3)

levels (e.g., lack of access to classroom spaces, microaggressions in classroom interactions; Dávila, 2015) impact student experiences with a particular focus on multimodal discourses. Thus, the purpose of this study was to better understand how girls of color labeled with significant cognitive disabilities in middle school and high school navigated multimodal discourses and classroom practices as well as how they were impacted by them. Two questions guided the inquiry: (a) How do girls of color labeled with significant cognitive disabili- ties multimodally navigate classroom discourses? (b) How do classroom multimodal discourses impact girls of color labeled with significant cognitive disabilities?

Method

This project focuses on a subset of data from a study examining educational opportunities for girls of color labeled with significant cognitive disabilities in one middle school and one high school in a large Midwestern city school district (see Miller, 2019). Like the larger study, this project used a critical, qualitative multiple case study design (Merriam & Tisdell, 2015).

Strategy of Inquiry

A multiple case study design was chosen for three reasons. First, the research questions required an in-depth understanding of how the focal participants navigated multimodal discourses and classroom practices as well as how they were impacted by them (Bhattacharya, 2017). Second, the research questions were explanatory, pro- cess-oriented, and framed as “how” questions. This allowed me to examine systems and processes while focus- ing on talk and action within learning contexts. Third, multiple case study methodology can offer new understandings inductively as bounded by a case and revealed across multiple cases (Merriam & Tisdell, 2015).

Conceptual Framework

This empirical project was grounded in Disability Critical Race Theory (DisCrit; Annamma et al., 2013) and critical discourse theory (Foucault, 1972; Gee, 2014; Rogers, 2011). First, DisCrit, as the broader theo- retical framework, is discussed. Then, critical discourse theory is described. Finally, a consideration of how the two theories strengthen one another is presented.

Disability critical race theory. A sibling of critical race theory and disability studies in education, DisCrit seeks to uncover how interlocking oppressions (e.g., ableism, racism) operate as institutional and societal mechanisms to oppress, segregate, and surveil multiply marginalized youth (e.g., youth of color with dis- abilities; Annamma et al., 2013). Each of DisCrit’s seven tenets affords an examination of how power is (re)produced in education (see Table 1). As an intersectional theory, DisCrit is particularly interested in human responses to difference and the activism and resistance individuals and groups engage in (Annamma et al., 2018). The question is not whether differences are perceived but what meaning is wielded based on those perceptions and the ensuing mutually constituted ideologies and consequences (Gallagher, 2001).

By affirming how racism and ableism are active and naturalized, DisCrit exposes how girls of color labeled with significant cognitive disabilities are outside perceptions of what is normal and thus are posi- tioned as problematic (Erevelles et al., 2006). Moreover, DisCrit questions the ways in which other, less prominent identity markers (e.g., communication preference, immigration status) may also be used to posi- tion students as different. For example, linguicism (Phillipson & Skutnabb-Kangas, 1996) may be used with students of color labeled with significant cognitive disabilities who use varied modes of action-oriented expression, low- and/or high-tech communication supports, or whose home language is not solely English. To do so, DisCrit centers the lived experiences and perspectives of multiply marginalized individuals and communities who are often unrecognized in research (Collins, 2003; Matsuda, 1987). It also considers the historical and legal aspects of disability and race and how both have been used independently and collec- tively to deny rights to some individuals (Gotanda, 1991). Such implications include how whiteness and ability are used as property wherein economic, political, and social benefits are afforded to those who are constructed as “white” while benefits are withheld from those who cannot claim whiteness (Harris, 1993).

Miller 161

Critical discourse theory. Foucault’s (1972) theory of discourse acknowledges how individuals make mean- ing of the social world through communicative acts. Multimodal discourses symbolize active and contex- tual processes that hold power and shape realities (Foucault, 1982). Since schools reproduce social realities, teachers and students play a role in how multimodal discourses and environments are produced (Rogers, 2011). Drawing on Foucault, I examined how particular ideologies (e.g., students are knowledge- able, disability is less than, talk is more important than action) were embedded in classroom multimodal discourses. Therefore, critical discourse theory strengthened DisCrit by affording an explicit examination of how linguicism operated in learning contexts, including how classroom meaning depended on how the focal participants were positioned, the opportunities provided to them, and the discursive moves and choices they made because of those affordances and constraints.

By grounding this work in DisCrit and critical discourse theory, I used multimodal discourses as analyti- cal tools to expose how intersecting oppressions at macrosociopolitical and microinteractional levels impacted the school experiences of girls of color labeled with significant cognitive disabilities. Talk and actions were examined to make more distinct the structures that are positioning these girls as invisible (Gee, 2014). That way, scholars and educators can address the negative impact certain systems and processes have on girls of color labeled with significant cognitive disabilities while showing students’ discursive strengths and educators’ acts of resistance.

Participants

Girls of color labeled with significant cognitive disabilities were purposively sampled aligning with the aims of the study (Lincoln & Guba, 1985). The phrase “labeled with significant cognitive disabilities” is used throughout this manuscript to (a) foreground the sociopolitical significance of disability and the preoccupation of medicine, psychology, and education with categorizing students as deficient (Connor & Gabel, 2010), particularly youth of color (Kulkarni, 2020), and (b) honor the journal’s person-first lan- guage requirement. Students were included in this analysis if they self-identified or their family identi- fied them as having autism, intellectual disability, and/or multiple disabilities. Six students were the focal participants. The school district designated intellectual disability and speech/language impairment labels on five of the students’ Individualized Education Programs (IEPs). Educators were also purposively sampled as secondary participants. At least one teacher was invited as a secondary participant per case. Seven teachers and one paraprofessional were included in the analysis. All participant names used in this article are pseudonyms. Student demographic information was taken from student and family demo- graphic questionnaires (see Table 2).

Data Collection and Sources

Three phases of data collection were conducted. This process allowed for the data to be collected and ana- lyzed iteratively as each preceding phase informed subsequent phase(s). For example, I collected observa- tion data, analyzed it for emerging themes, turned hunches into questions, and returned for another

Table 1. Tenets of Disability Critical Race Theory (DisCrit).

DisCrit tenets

Tenet One uncovers how racism and ableism circulate interdependently. Tenet Two values multidimensional identities. Tenet Three emphasizes the social constructions of ability and race. Tenet Four privileges the voices of multiply marginalized individuals and groups. Tenet Five considers ideological, historical, and aspects of disability and race. Tenet Six examines how ability and whiteness operate as property. Tenet Seven necessitates activism and upholds all forms of resistance.

162 Research and Practice for Persons with Severe Disabilities 47(3)

observation with new questions (Bhattacharya, 2017). Similarly, each focus group was a chance to continue ongoing member checks with focal participants (Rodwell, 1998), as the information gathered from the first and second interviews iteratively informed follow-up questions for the focus groups.

Primary data sources. Classroom observations were primary data sources. I conducted 3-5 observations of teaching and learning in general and special education classrooms, including audio/video recordings and detailed field notes. This yielded 28 audio/video recordings and 27 detailed field notes as primary data sources. Field notes from one observation were missing during the analysis. Four focal participants were afforded access to only general education special or elective classes (e.g., Choir, Theater). Two students (Amy and Luna) were only assigned to one segregated special education classroom across the school day, so observations during community-based instruction were also conducted.

Secondary data sources. Interviews and focus groups were secondary data sources. Each focal participant had 2 to 3 interviews and 1 to 2 focus groups, yielding 17 student interviews, 2 high school focus groups, and 1 middle school focus group. The girls and their families also completed demographic forms. In three cases, parents shared additional information via phone and in-person about their daughter’s educational trajectory with me. Seven teachers and one paraprofessional consented to the observation component of the study. Four teachers participated in two interviews and two teachers participated in one interview. Mr. Armstrong, one of the general education teachers, and Ms. Cari, the paraprofessional, did not participate in the interview com- ponent due to scheduling and time constraints. This yielded 10 teacher interviews. Teacher interviews existed across more than one case because most focal participants were in the same segregated classrooms (e.g., Meena, Emma-Mae, and Isabella, Amy and Luna). Secondary sources contextualized observations and trian- gulated data (Brantlinger et al., 2005).

Primary Data Source Preparation

First, the audio/video recordings of teaching and learning were turned into verbatim transcriptions of talk- by-speaker utterances as an entry point to data analysis (Gee, 2014). Utterances were defined as completed words, partial words, and on-record vocalizations within each observation. Transcribing three types of utterances helped answer both research questions because they revealed how students and teachers com- municated with one another. In addition, they showed the importance of vocalizations to students without

Table 2. Participants and Classroom Content Areas by Case.

Student participant Age Grade Race/ethnicity Disability label

Educator participant

Content area observations

Amy 16 11th Hispanic Down syndrome; SLI

Mr. Clifford Ms. Cari (Para)

Language Arts (SE) CBI (SE)

Emma-Mae 11 6th Black ADHD; ASD Ms. Taub Ms. Snow Ms. Summitt

Language Arts (SE) Physical Education (GE)

Isabella 14 8th Latina ASD Ms. Snow Mr. Fenn

Social Studies (SE) Theater (GE)

Jimena 19 1st year post-secondary

Hispanic OHI; OI; SLI; VI/B

Ms. Parker Mr. Armstrong

Language Arts (SE) Choir (GE)

Luna 14 9th Latina Epilepsy; ID; SLD; TBI

Mr. Clifford Ms. Cari (Para)

Language Arts (SE) CBI (SE)

Meena 14 8th Afghan OI; SLI Ms. Taub Mr. Fenn

Language Arts (SE) Theater (GE)

Note. SLI = speech or language impairment; SE = special education; CBI = community-based instruction; ADHD = attention deficit hyperactivity disorder; ASD = Autism spectrum disorder; GE = general education; OHI = other health impairment; OI = orthopedic impairment; VI/B = vision impairment/ blindness; ID = intellectual disability; SLD = specific learning disability; TBI = traumatic brain injury; Para = paraprofessional.

Miller 163

communication supports, how often interruptions without repairs occurred, and how prevalent educators responded to talk but not actions. Then, details were iteratively added (e.g., tonal marks, truncated or inter- rupted speech, and pauses were noted; Ochs, 1979).

Talk was then organized by lines based on speaker intonation, action, and interruption. Some lines had only one utterance while others had multiple utterances. Interruptions were shown with a code rather than dictated by the placement of the utterance on a line (Du Bois, 2006). Next, lines were organized by stan- zas—a group of lines about a theme, happening, or topic (Gee, 2014). Lines were organized by stanzas based on turn sequences and topics. A new stanza began when the teacher started talking to the whole class or when topics shifted. It was important to organize lines in this way to understand classroom participation structures and teacher and student initiations and responses (Cazden, 2001).

Next, detailed descriptions of salient actions were added to the transcripts (Norris, 2004). Because I was most interested in the focal participants’ classroom experiences, I focused on actions they used most often, including eye gazes, facial expressions, body movements, and gestures. Like talk, actions were indicated line-by-line as speakers took turns. When there was simultaneous talk and action, interruptions are shown with a code rather than dictated by placement on a line (see Table 3 for transcription conventions).

Researcher Positionality

As a White, cisgender woman who identifies with several nonvisible disabilities, the focal participants and I did not share age, disability label, race, or ethnicity. However, the participants and I did share gen- der and language. Thus, gender and language were the starting points of commonality that we built trust from. I volunteered in their special education and general education classrooms for several weeks before the project started. Before, during, and after data collection, I spent time with the girls across the school day, including during class, at lunch, and on class trips. Also, I took extra steps to ensure the project was enjoyable and generative for them. Ensuring a generative project meant thinking with and alongside the focal participants about the problems they were facing in school and focusing on their solutions to ineq- uitable school systems and processes (Kinloch & San Pedro, 2014). In addition, and as part of solution- generating, I examined multimodal discourses with the educators who participated in second interviews to reflect on their teaching and make important pedagogical changes focused on girls of color labeled with significant cognitive disabilities.

Ableism, linguicism, and racism held real consequences for the focal participants that I did not experi- ence. For example, I moved around both schools with relative ease. At the high school, I learned and used the code to get in and out of “the annex” freely (the separate building at the high school for the segregated special education classes), whereas the high school girls could not come and go unless they were with me or a school staff member. At both schools, I used the dominant mode of expression which granted me positioning and privileges not afforded to the focal participants. Through reflexive journaling and peer debriefing, I continu- ously considered how my analysis of classroom discourses and my presence could impact the students.

Table 3. Transcription Conventions.

Code Meaning Example

. Final tone, a period in speech Emma-Mae: Ms. Taub. Ms. Taub. , A non-final tone, like a comma in speech Isabella: Oh wait, wait. No, no. ? A rise in pitch, like a question in speech Mr. Clifford: Amy, do you want to bowl? ! A rise in pitch, like an exclamation in speech Luna: I did it myself! – Truncated, cut-off word/interrupted speech Isabella: Um, the ma:: mash::- – Truncated, cut-off action/interrupted action Jimena: [Pointing and looking ahead, in that same direction-] [ ] Student and teacher actions Meena: [Looking at Ms. Taub. Raises her hand again.] :: Lengthening of syllable Mr. Fenn: There are winners in Wah:: () Time passed in seconds (12)

164 Research and Practice for Persons with Severe Disabilities 47(3)

Data Analysis

The data analytic plan was informed by the study purpose, conceptual framework, and strategy of inquiry. Critical multimodal discourse analysis (Kress, 2010) was used to develop codes of multimodal discourses within cases and then across cases following data analysis for multiple case study design (Merriam, 2001). Once all data had been collected, iterative data analysis continued as I searched for patterns across the data (Erickson, 1986). I moved back and forth reading the data, turning hunches into questions or writing down new questions, and returning to the data to look for patterns (Bhattacharya, 2017). As such, I engaged in multiple rounds of meaning-making and coding.

First cycle: Within-case analysis. During within-case analysis, I used inductive analysis, open to ideas that emerged from the data not yet represented in the literature (Erickson, 1986). I moved through three rounds of initial coding, including unitizing, categorizing, and labeling (Rodwell, 1998) attentive specifically to the focal participants’ multimodal discourses (e.g., Answers Teacher’s Question and Initiates About Finished Work both emerged as inductive categories). Then, the deductive analysis focused on how the talk was structured and the roles of actions. Using this process, patterns in the data were identified based on the lit- erature (e.g., student repositioning) and the conceptual framework (Annamma, 2018; Collins, 2011a e.g., Repositions was a deductive code category).

Second cycle: Across-case analysis. During across-case analysis, I used axial coding (Rodwell, 1998), placing code categories across the six cases in relation to one another and used data displays to compare and cluster data across cases. Clustering and comparing were helpful when considering similarities and differences across cases. The checklist matrix data display helped me expand and collapse code categories (Miles & Huberman, 1994). For example, I expanded the code category Responds to Teacher (Not Answering a Question) as it became important to draw out when the focal participants were responding to teachers. Expanding this code category highlighted the ways in which discourse was controlled in the classroom, including in student responses. Then, I asked new questions about how that discursive control was impacting the positioning or importance of talk over action in the classroom. I engaged in three cycles of inductive analysis across cases.

Finally, I engaged in one cycle of deductive analysis wherein the conceptual framework was used to look for intersecting oppressions the students experienced while remaining open to those that arose from the data. For example, layers of linguicism were exposed as schools failed to recognize and embrace the focal participants’ multilingual strengths (Mindel & John, 2018; Young, 2009). These practices were revealed within the code categories of Expresses a Need or Want and Asks a Question.

Trustworthiness

Several strategies to support the trustworthiness of the findings (Lincoln & Guba, 1985) and advance rigor (Bhattacharya, 2017) were used including iterative data collection and analysis, disconfirming evidence, and analytic memos (Miller, 2020; Miller & Kurth, 2021). For brevity, triangulation and reflexive journaling are discussed next.

Triangulation improved the probability that the findings and interpretations were credible and trustwor- thy. In this study, data and methodological triangulation were used. First, data were collected from more than one participant source (e.g., students and teachers). Second, several data collection methods were used, including field notes and transcriptions of classroom discourse as primary data sources and interviews and focus groups as secondary data sources. Using data and methodological triangulation allowed me to look for patterns and outliers across sources and contexts.

Reflexive journaling supported trustworthiness as it revealed my orientation toward inquiry, social action, and analysis (Saldaña, 2013; e.g., I wrote in a research journal to interrogate reactions, check work- ing hypotheses, and generate novel ideas). During the analysis, I journaled to remain close to the data and existing literature months after data collection. Also, journaling was used to examine how my participation in the study contributed to the production or disruption of power. In summary, journaling was a way to honor axiological commitments and understand project roles.

Miller 165

Results

Next, I share two themes that emerged from the analysis focused on how girls of color labeled with signifi- cant cognitive disabilities engaged in multimodal discourses with educators.

Students’ Resourceful Multimodal Discourses

Across cases, girls of color labeled with significant cognitive disabilities used resourceful multimodal dis- courses in the classroom. Their discourses were resourceful because of how the focal participants conver- sationally navigated their classrooms so they would be honored and strengthened, not discounted or remediated. Students’ Multimodal Discourses was defined as “Focal participants communicate with their teachers about their learning through talk and actions.”

At the middle school. It was Friday and the middle school students were graphing their scores for the whole group Daily Language Review activities (e.g., sentence structure, grammar). Emma-Mae used talk and action to inquire about her missing scores. She initiated with Ms. Taub for help (line I) and the teacher responded with eye contact (line I.1) and a question (line I.3). However, when Emma-Mae encouraged Ms. Taub to look at her paper as a discursive move to support her in conveying her thoughts (line I.6), Ms. Taub did not walk over to Emma-Mae and engage with her. Instead, she told Emma-Mae that she would look at her packet later (line I.7, line I.9):

I. Emma-Mae: Ms. Taub. Ms. Taub. [Looking at Ms. Taub.] 1. Ms. Taub: [Turns her eyes away from another student and looks at Emma-Mae.] 2. Emma-Mae: I don’t have any numbers. [Looking down at her paper then at Ms. Taub from the side.] 3. Ms. Taub: Oh, for Wednesday and Thursday, you guys didn’t grade them? 4. Emma-Mae: Yeah. 5. Ms. Taub: That’s fine. 6. Emma-Mae: See look. [Picks the paper up from the table, turns sideways in her chair. Looks at and

points to her paper with her pencil. Looks at Ms. Taub.] This one doesn’t have any numbers. 7. Ms. Taub: [Looks away from another student and back at Emma-Mae.] I’ll just look at it. 8. Emma-Mae: [Turns in her chair and faces the front again. Continues pointing at her paper with her

pencil while looking at Ms. Taub.] So, I don’t have anything for Wednesday because we don’t have any numbers.

9. Ms. Taub: [Looking at Emma-Mae.] I’ll look at ’em, ok? [Looks at the whole class.] 10. Emma-Mae: [Looks at her paper.]

Emma-Mae held subtle discursive power as she tried to get help from Ms. Taub. However, her academic earnestness was diminished when the teacher would not look at her paper. Their interaction ended when Ms. Taub started giving directions to the whole class.

During an audio-recorded Social Studies lesson missing some video footage, Isabella responded to Ms. Snow’s anticipatory query. Initially, Ms. Snow used closed or known-answer questioning (a technique wherein the teacher already has an answer in mind) with the class to prepare for a short video on New Hampshire (line II). Importantly, students hold limited power when known-answer questions are present. Still, Isabella attempted a response (line II.1) but because she expected a certain response, Ms. Snow inter- rupted Isabella with an additional prompt (line II.2). In response, Isabella processed aloud and repaired (line II. 4). Then, she answered Ms. Snow’s known-answer question two turns later (line II.6). Afterwards, Ms. Snow praised her (line II.7):

II. Ms. Snow: The largest city, thank you. Isabella, what else are we going to be looking for? 1. Isabella: Um, the ma:: mash::- 2. Ms. Snow: No, on your, your book.

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3. Student: Qué libro? 4. Isabella: Where am I at? Oh wait, wait. No, no. 5. Ms. Snow: Turn the other way. No, what are we going to be looking for on the next page here?

[Points to the page in her book.] On this page, babe. [Taps the page in the book.] Someone’s already said nickname. Someone’s already said largest city. What else? Pick one, Isabella.

6. Isabella: Um. Um. Um. Um. Um. Um, landmark of the state. 7. Ms. Snow: Landmark, good. What else? (Student), what else are we going to listen for?

Teachers used known-answer questions often. As such, Isabella received praise from Ms. Snow when she responded correctly. If Ms. Snow had tried to repair with Isabella after she interrupted her (e.g., asking more about what Isabella was thinking or had meant to say), then her discursive move would have honored Isabella as a thinker and a learner beyond having the “right” answer.

At the middle school, the class was working independently decorating their journal pages with drawings of holiday-themed objects and characters after a writing lesson about Halloween jokes. Meena used actions to connect with Ms. Taub about her finished work. In her third interview, Meena shared that writing for extensive periods of time made her hand feel sore. She also indicated this through her actions (line III). Here, Meena tried more than once (line III, III.1, III.2, line III.4) to get her teacher’s attention. After four tries, Ms. Taub noticed Meena (line III.5) and permitted her to come to the front of the room through her actions:

III. Meena: [Looks up from her journal at Ms. Taub sitting at the front of the room. Raises her hand and arm briefly, then puts it down. Shakes her left hand in the air. Looks down at her journal and then up at the big cup of coloring pencils. Picks up the big cup and returns it to the art shelf.] (10) 1. Meena: [Sits back down in her seat. Looks at Ms. Taub while she picks up her notebook, holds it

with two hands close to her. Turns her body to face Ms. Taub. Looks at and watches Ms. Taub talk to Emma-Mae.]

(5) 2. Meena: [Looking at Ms. Taub. Raises her hand and then puts it down.] 3. Ms. Taub: [Looks up from the table to the right side of the room.] 4. Meena: [Looking at Ms. Taub. Raises her hand again.] 5. Ms. Taub: [Scans the room toward the left and looks at Meena raising her hand. Smiles. Looks down

at Emma Mae’s notebook. Motions with her left hand for Meena to come to the front of the room.] 6. Meena: [Stands up. Pushes in her chair. Walks to the front. Sets her notebook down in front of Ms.

Taub who is talking to Emma-Mae. Looks at Ms. Taub.]

Meena showed discursive ingenuity by using multiple actions (e.g., gestures, eye contact, body movements) to initiate with Ms. Taub about her finished work. Moreover, she did so over time while also waiting until the teacher looked up from the conversation that she was engaged in.

At the high school. In Luna’s case, she used talk to initiate about her Language Arts work. While there is some video footage missing in this example, Luna shared how she had finished her Esperanza Rising col- lage (line IV). When no one responded, she pronounced completion again (line IV.1) with an emphasis that she had worked independently, a common school goal, particularly for students receiving special education supports and services. When there was still no response, Luna directed her comment to a paraprofessional (line IV.2) who took up her initiation and praised her with talk and actions (line IV.3):

IV. Luna: I did it myself! I did it. I did it! (8) 1. Luna: All by myself! (12) 2. Luna: Cari, I did it myself.

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3. Ms. Cari: Alright! [Gives Luna a high five.] Good job! Put your name up at the top so we make sure it’s your paper. Oh, let me find you a pencil.

4. Luna: [Turns to talk to a classmate.]

In a crowded segregated classroom, this was inherently a resourceful discursive move by Luna to gain adult recognition of her completed work. However, no one discussed her college with her, and the opportunity to engage with Luna about her learning on a deeper level was missed.

In her second interview, Jimena shared that she liked reading the Twilight series and preferred the second book. Before each chapter, Ms. Parker asked the students to make a prediction about the outcome. This time, she asked, “What causes chaos at Bella’s birthday celebration at the Cullen’s? Is it an ex-girlfriend? Or blood?” Jimena used actions to communicate her literary prediction. When Ms. Parker approached Jimena, she repeated the two choices (line V). Jimena communicated her prediction with gestures (line V.1). When Ms. Parker confirmed (line V.2), Jimena used additional multimodal discourses, including eye con- tact, head movements, and facial expressions (line V.3) to ensure she was understood. Ms. Parker responded with talk and actions affirming Jimena’s choice (line V.4):

V. Ms. Parker: Jimena, what do you think? Blood or an ex-girlfriend? [Holds out two visual choices on little pieces of paper, one in each hand. Ex-girlfriend and blood are depicted with pictures from Boardmaker.] 1. Jimena: [Looks at the visuals. Reaches out and selects blood with her right hand.] 2. Ms. Parker: You’re going blood too? 3. Jimena: [Looks at Ms. Parker. Nods head yes. Smiles.] 4. Ms. Parker: Ok. [Nods her head. Sets the two choices down on the table.]

Ms. Parker always presented the students with two predetermined choices for their predictions. Here, she did not engage deeply with Jimena about why she made that prediction or what it meant for the broader storyline. Then, after gathering everyone’s predictions, Ms. Parker read the chapter aloud without pauses, guided questions, or conversation. Afterward, Ms. Parker asked each student individually to confirm if their prediction was correct.

Across cases, there were instances when teachers did not respond to or missed the girls’ initiations. For example, Amy used talk five distinct times over the course of more than 6 min to initiate a turn with Mr. Clifford about her completed project in Language Arts. Despite Amy’s enthusiasm and persistence, neither Mr. Clifford nor the paraprofessionals in the room responded to her. It was possible that Mr. Clifford did not see or hear Amy as she did not approach him but remained seated at her desk. However, Amy’s discursive opportunity lost all potential when the adults (the intended audience) did not respond. Girls of color labeled with significant cognitive disabilities held subtle discursive power when teachers did respond to their initiations. Consequently, they held no discursive power when teachers did not respond.

Students’ Strategic Repositioning

Across cases, focal participants repositioned or refused to accept marginalization. I expand on prior defini- tions (Annamma et al., 2020; Miller et al., 2022) to define Student Repositioning as, “Girls of color labeled with significant cognitive disabilities engage in strategic maneuvering in response to individual, school, or societal marginalization.”

At the middle school. Girls of color labeled with significant cognitive disabilities repositioned by mentioning their strengths and skills. For example, Isabella repositioned in response to Mr. Fenn, the Theater teacher, in the general education classroom. As the students transitioned to a new game, Mr. Fenn commented on the room’s temperature and how that should not impact participation (line VI). In that same turn, he also prompted Isabella and a classmate to partake in the activity. Isabella repositioned by stating that she was good at the game (line VI.2). However, Mr. Fenn ignored Isabella’s comment (line VI.3):

168 Research and Practice for Persons with Severe Disabilities 47(3)

VI. Mr. Fenn: Even though it’s too cold, stand up. So, our hands are here. First step. Show me your hands, (Student). We need hands for the game even though you’re cold, put your hands up. Isabella, let’s make these hands. Let’s go. 1. Student: I’m good at this. 2. Isabella: Me too. 3. Mr. Fenn: Hey (Student), can you find that space where you’re facing us not out the way? Here we

go. This is the least engaging part of this activity. Let’s get the instructions and then we can go. It’s an elimination-based game. There are winners in Wah:: Pay attention so you can be a winner.

The power that focal participants could hold when their teachers ignored their repositioning was context dependent. In this instance, Isabella gained no discursive power when Mr. Fenn did not respond to her. Instead, Mr. Fenn could have affirmed Isabella’s comment about her skills.

Emma-Mae used physical maneuvering to reposition at the literacy station. After losing the opportunity to sit in a seat of her choice (line VII) and being told how to sit (line VII.2), Emma-Mae repositioned and responded with a personal choice—using her pencil to point to the first word instead of her finger (line VII.5). Instead of allowing her this subtle personal choice, Ms. Snow directed Emma-Mae to use her finger (line VII.6) and Emma-Mae followed the direction (line VII.7):

VII. Ms. Snow: I’ll still help you bud. [Looking down at the table, pulls out the chair next to her.] I need her to sit here. [Pats the table.] 1. Emma-Mae: [Sits down next to Ms. Snow with her spelling journal and pencil. Looks at Ms. Snow’s

journal.] 2. Ms. Snow: Ok. Feet on the floor? [Looks down at Emma-Mae’s feet.] 3. Emma-Mae: [Looks past the peer across the table. Puts her foot that was crossed under her down on

the floor.] 4. Ms. Snow: Great. Here we go. [Looking down at her own spelling journal.] Put your finger on the

first word. [Looks over and down at Emma-Mae’s journal.] 5. Emma-Mae: [Looking down at her journal, points to the first word with her pencil in her left hand.] 6. Ms. Snow: Finger. 7. Emma-Mae: [Puts the pencil down. Still looking down at her journal, points to the first word with

her right pointer finger.]

In this instance, Ms. Snow used directions and redirections to respond to Emma-Mae’s repositioning. Student repositioning held no power when the teacher would not affirm or allow it.

At the high school. Luna used talk to reposition regarding her academic participation in Language Arts. As Mr. Clifford transitioned to the next activity (line VIII), Luna used talk to interrupt and speak out in response to the notion that not everyone was going to be involved (line VIII.1). Similar to other activities, this one focused on the story’s plot and was not age sensitive or differentiated. Yet, during the first focus group, Luna expressed how much she enjoyed Esperanza Rising. Mr. Clifford responded with a general reminder (line VIII.2) without engaging with Luna:

VIII. Mr. Clifford: So, if we have everybody’s collage, we’re moving on here. Kinda look at what, what’s, to look at what’s next. We’re going to, a lot of us are going to work on this project. I’m just going to kinda, that is also related to our book. Not everybody’s going- 1. Luna: Me too. 2. Mr. Clifford: I want everybody listening even though I’ll get it to you. If it’s, basically what you

have here is a flip book. We kind of show the beginning of a book with your drawings. Sort of the beginning. . .

Speaking out was one strategy focal participants used to reposition in the classroom. Teachers’ responses (e.g., ignoring, redirecting, taking up) dictated how much (if any) discursive power was afforded to focal participants when they multimodally spoke out.

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Focal participants also repositioned by honoring their needs and wants. After cleaning the arcade during community-based instruction, sometimes the students got to go bowling. It was the only part of the arcade that was open for play. When Amy arrived, her peers had already started. She sat down on one of the nearby cushioned benches without any materials. When Mr. Clifford asked her if she wanted to bowl (line IX), Amy repositioned and responded to Mr. Clifford by pairing actions (i.e., moving her head and pointing) with eye contact (line IX.1). Her message was clear as seen in Mr. Clifford’s response (line IX.2):

IX. Mr. Clifford: Amy, do you want to bowl? Do you want to bowl? There’s a lane over there. 1. Amy: [Looks at Mr. Clifford. Shakes her head no and then points to the seat cushion she is sitting

on.] 2. Mr. Clifford: Nope, no. Ok.

Although he did not inquire further, Mr. Clifford afforded Amy subtle discursive power by acknowledging and responding to her. Opting out was one way Amy could hold some power at the moment.

Focal participants also repaired or attempted to repair when misunderstood (Macbeth, 2004). During the transition from Language Arts to the next activity, Jimena initiated a request with Ms. Parker through talk and action (line X). In response, Ms. Parker asked similar questions while using context clues (line X.1, line X.3, line X.5). Yet, she did not vary her questions. When Ms. Parker misunderstood, Jimena physically repositioned by using gestures and eye gaze to repair (line X.2, line X.4, line X.6). After these three addi- tional turns, Jimena was understood, and her request was approved (line X.7):

X. Jimena: Eh. [Pointing ahead of herself to the other side of the room and looking in that direction.] 1. Ms. Parker: [Walking away and looking toward the class.] Huh? [Stops and looks at Jimena.] What

did I forget? Your foot things? 2. Jimena: [Pointing and looking ahead.] 3. Ms. Parker: What? [Looks in the direction Jimena points.] 4. Jimena: [Turns to look at her. Taps Ms. Parker. Turns and points ahead of her again.] 5. Ms. Parker: What? [Looks in the direction Jimena points.] 6. Jimena: [Pointing and looking ahead, in the same direction-] 7. Ms. Parker: Oh. [Points in the same direction as Jimena.] You can go see Judy if you want to. 8. Jimena: [Smiles. Looks ahead. Reverses her wheelchair and then starts moving toward Judy.]

During her second interview, Ms. Parker described how she had not tried low- or high-tech communication supports this school year because “most of the time, it really boils down to knowing (Jimena).” It was more important to know Jimena because the district provided “the same, like, solution for every kid” rather than person-centered supports. As such, Jimena’s repositioning through repair was also a response to the lack of supports.

Discussion

Girls of color labeled with significant cognitive disabilities initiated with and responded to their teachers about their ongoing and completed work through multimodal discourses. They asked questions about their learning (e.g., for help) and made requests (e.g., permission). As DisCrit tenet one foregrounds, racism and ableism position girls of color labeled with significant cognitive disabilities as less than and not capable (Annamma et al., 2013). Therefore, it may be assumed that they cannot share information, ask questions, and make requests. However, the findings here are consistent with prior research in that youth claimed nuanced discursive power by asking questions (Ingram & Elliott, 2014) and sharing information (Brooks, 2015). The findings extend prior research showing how girls of color labeled with significant cognitive dis- abilities also claim discursive power through questioning and sharing information.

Another contribution of this study, supported by DisCrit tenet seven (Annamma et al., 2013), centers the focal participants’ repositioning through various discursive strategies as creative acts of resistance. The

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findings described here support existing research examining how students reposition by speaking out (Annamma et al., 2020) and sharing personal information (Collins, 2011b). This study expands that litera- ture with a particular focus on the strategies the focal participants employed including physical maneuver- ing, repairing, and opting out. Little is known about how girls of color labeled with significant cognitive disabilities reposition or refuse individual, classroom, and societal marginalization and how their teachers respond to them. This study presents novel information for future scholarship to build upon.

This study demonstrates how girls of color labeled with significant cognitive disabilities had few visible supports for communicating with their teachers. Here, DisCrit tenets one and six blended with critical dis- course theory illuminated how racism and ableism intersected with linguicism and multimodal communica- tion supports, technologies, and tools were withheld from the focal participants. As a result, they had to work hard to communicate and express themselves across the school day, holding the burden of presenting themselves as knowledge generators (Delgado Bernal, 2002). While most of the girls often successfully dialogued without supports, exemplifying their agency and resourcefulness in the classroom (Tichavakunda, 2021), their discursive strengths and gifts were not responded to.

Educators responded to the focal participants with directions and redirections, questions, and praise. At times, they interrupted or made assumptions about students’ discourses. This finding supports existing research wherein educators’ interruptions disrupted the fluency of the initiation and subtracted from the subtle discursive power youth could claim (Bliss et al., 1998). At times, teachers made repairs to ensure understanding and other times they did not. Also, some teachers did not help the students when they asked for help (Annamma et al., 2020). These findings add to the literature by considering the kinds of unproduc- tive interactions girls of color labeled with significant cognitive disabilities had with educators.

Teachers did not vary their questioning techniques as most questions were closed or known-answer ques- tions rather than open questions (Piccolo et al., 2008). In this study, praise and affirmation replaced open- questioning techniques, resulting in brief turn sequences and little to no opportunity to engage deeply with the content. Black (2004) found that teachers varied their questioning based on how they perceive students’ abilities, resulting in more controlling forms of talk when teachers’ perceptions of students’ abilities were deficit-based. In this study, teachers’ responses demonstrated how students were positioned. This position- ing resulted in teachers withholding (a) goodness and smartness and (b) meaningful learning from girls of color labeled with significant cognitive disabilities, as identified through DisCrit tenet six (Leonardo & Broderick, 2011).

Importantly, there were instances wherein teachers did not respond to the focal participants (Clarke, 2007), resulting in no shifts in discursive control. At times, this may have been because the teacher did not see or hear a student as the girls did not always physically approach teachers or say a teacher’s name before initiating. In crowded special education classrooms, educators were often in proximity when communicat- ing with a focal participant. Moreover, this omission highlights how classrooms prioritize oral/aural com- munication over actions. The findings presented here add to the literature as teachers most often controlled classroom discourses and upheld talk as the dominant mode of expression, thus limiting opportunities for the girls to play more active learning roles multimodally.

Limitations and Future Research Directions

While the focus on students as primary participants is a strength of the study, the lack of stakeholders beyond students and educators is a limitation. Therefore, one research implication revolves around partici- pation and supports the focus of DisCrit tenet two on participants’ multidimensional identities. For exam- ple, speech-language pathologists would provide insight into IEP team collaboration continuities and fissures when considering communication supports and tools (Soto & Yu, 2014). Inviting participation from additional school personnel, including school administrators, would be an important next step while still centering girls of color labeled with significant cognitive disabilities.

Although student-generated data and student-involved analysis are also strengths of the study, more youth participatory action research (Cammarota & Fine, 2008) led by girls of color labeled with significant cognitive disabilities is needed. Such scholarship would (a) be grounded in the girls’ lived experiences and

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concerns, (b) incorporate training and practice of research skills, (c) iteratively combine research and action, and (d) involve power-sharing between adults and youth. Youth participatory action research is a promising tool for engaging girls of color labeled with significant cognitive disabilities in youth-driven transformative educational change.

Reimagining Schooling

The first implication for reimagining schooling focuses on how girls of color labeled with significant cogni- tive disabilities are positioned and supported in schools. In this study, some teachers supported the girls’ multimodal discourses by getting to know them and what they needed. This was often done from the per- spective of student learning need (Naraian, 2016) rather than from the perspective that these students have strengths and gifts (Annamma & Morrison, 2018). While educators resisted the school’s marginalizing practices in these microinteractional ways, their efforts were often spent initiating communication instead of findings ways for classroom discourses to be multidirectional and multimodal. In this way, interactions felt one-sided with the adult holding the power. Instead, girls of color labeled with significant cognitive disabilities must be positioned as epistemic agents (Taylor & McDonough, 2021) and vulnerable assets (Proffitt, 2020) rather than solely as knowledge consumers, and supported as such.

A second implication focuses on the necessity to expand what is considered discourse. In schools, educa- tors often prioritize oral/aural communication over actions. Yet, communication is richer and more complex than that. People often use multimodal discourses across contexts and communication partners (Jewitt, 2017). Expanding what is considered discourse by positioning actions as important as talk is essential for all students, particularly students labeled with significant cognitive disabilities and students with complex communication needs.

A third implication centers on multimodal communication supports, technologies, and tools as an inte- gral part of schooling (Hamraie, 2013). This includes providing access to and use of low-tech (e.g., com- munication book, eye-gaze board) and more complex systems, sign language, and multiple languages. Here, girls of color labeled with significant cognitive disabilities and their families are positioned as experts leading/co-leading multimodal discourse access considerations so that the design, development, and imple- mentation are “both accessible and responsive” (Foley & Ferri, 2012, p. 199), rather than tailored to district conveniences and/or school personnel comfort. Furthermore, optimal multimodal communication supports, technologies, and tools are person- and family-centered and change over time as necessary (Yu et al., 2021). In summary, girls of color labeled with significant cognitive disabilities ought to be afforded ample oppor- tunities to experiment with and use varied multimodal communication supports, technologies, and tools consistently, ones that incorporate their multilingualism authentically.

Conclusion

The purpose of this study was to expand current understandings of how multimodal discourses and class- room practices impacted youth with disabilities broadly by considering the unique intersectional experi- ences of six girls of color labeled with significant cognitive disabilities. Critical multimodal discourse revealed how the focal participants showed their discursive resourcefulness. Meaning, they used discursive strategies that should be honored and strengthened to navigate their classrooms despite absent communica- tion supports and prioritization of oral/aural communication. In addition, the focal participants repositioned in response to marginalization through talk and actions, including physical maneuvering, repairing, and opting out. Ultimately, this study underscores the necessity to center the experiences of girls of color labeled with significant cognitive disabilities in educational research to reimagine schooling.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

172 Research and Practice for Persons with Severe Disabilities 47(3)

Funding

The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the U.S. Department of Education (DED0072555).

ORCID iD

Amanda L. Miller https://orcid.org/0000-0002-5342-0717

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Author Biographies

Amanda L. Miller is an associate professor of education at Wayne State University. Her research focuses on youth perspectives, family-school-community partnerships, and disability-centered, culturally sustaining pedagogies.

Received: January 31, 2022 Final Acceptance: June 20, 2022 Editor in Charge: Sharon M. Darling

Copyright of Research & Practice for Persons with Severe Disabilities is the property of Sage Publications Inc. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.

Systematic Review

The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

G. Fynn,1 M. Porter,1 T. Borchard,1 C. Kazzi,1 Q. Zhong1 & L. Campbell2

1 School of Psychology, Macquarie University, Sydney, New South Wales, Australia 2 School of Psychology, University of Newcastle, Newcastle, New South Wales, Australia

Abstract

Introduction Individuals with intellectual disability (ID) are at higher risk of experiencing difficulties with anxiety than the general population. However, there are major barriers for individuals to receive appropriate services. There is a growing understanding of the importance of developing appropriate psychological interventions for this group. The objective of the current review was to systematically evaluate the findings of studies investigating the effectiveness of cognitive behavioural therapy (CBT) for individuals with ID and anxiety. Another aim was to explore which adaptions to CBT and treatment components were currently being utilised within the field. Method The electronic databases of CINAHL, EMBASE, Medline, PsycINFO, Psychology and Behavioural Sciences Collection and Scopus were searched to identify relevant studies. The methodological quality of these studies was assessed using established quality assessment tools by the

National Institutes of Health for pre and post studies and case series. Results Nine studies were included in this systematic review, all of which reported improvements in anxiety severity for some participants (25%–100%; N = 60) following CBT. Only three studies reported moderate effect sizes for CBT interventions on anxiety for individuals with ID. Discussion and Conclusions There is emerging literature supporting the effectiveness of CBT for individuals with mild ID. Findings highlight that CBT for individuals with anxiety and mild ID, including cognitive components, may be feasible and tolerable. While the field is gradually receiving more attention, there are significant methodological flaws present, which limit the conclusions that can be drawn regarding the effectiveness of CBT for individuals with ID. However, there is emerging evidence for techniques such as cognitive restructuring and thought replacement and modifications such as visual aids, modelling and smaller groups based on this review. Future research is warranted to investigate whether individuals with more severe ID can benefit from CBT, as well as further exploring what are the necessary components and modifications.

816

Correspondence:

A/Prof Melanie Porter, Room 3.519, AHH, School of Psychology,

Macquarie University, Herring Road, Sydney, New South Wales

Australia 2109.

(e-mail: [email protected])

Journal of Intellectual Disability Research doi: 10.1111/jir.13046

VOLUME 67 PART 9 pp 816–841 SEPTEMBER 2023

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

This is an open access article under the terms of the Creative Commons Attribution-NonCommercial License, which permits

use, distribution and reproduction in any medium, provided the original work is properly cited and is not used for commercial

purposes.

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Introduction

Individuals with an intellectual disability (ID) are at greater risk of experiencing mental illness at all ages, compared with the general population (21.7% vs. 4.3%; Hughes-McCormack et al. 2017). Nonetheless, anxiety is underdiagnosed in the ID population due to diagnostic challenges (Hsieh et al. 2020). First, people with ID are less able to self-report anxiety symptoms due to expressive communication difficulties (Scott & Havercamp 2014); second, anxiety disorders may wrongly be attributed to pre-existing ID (i.e. diagnostic overshadowing; Reiss et al. 1982). This lack of diagnostic sensitivity impedes appropriate treatment.

Individuals with an ID are at greater risk of developing anxiety due to various factors, such as an increased number of stressful life events or chronic health conditions (Hsieh et al. 2020), and they may have limited psychological resources to cope with adverse experiences (Taylor et al. 2008). Moreover, several neurodevelopmental conditions with comorbid ID are associated with elevated anxiety, such as autism spectrum disorder (ASD; van Steensel & Heeman 2017), Fragile X syndrome (Cordeiro et al. 2011) andWilliams syndrome (Kozel et al. 2021). Despite this, there is a lack of evidence-based approaches in clinical practice and limited expertise and confidence of professionals working with people with ID and concurrent mental health difficulties, which may contribute to the difficulties accessing appropriate services (Unwin et al. 2016).

Historically, there has been an overreliance on medication and behaviour modification when treating mental health difficulties of individuals with an ID (Whitehouse et al. 2006). It was commonly asserted that psychological therapy involving talk therapy or cognitive components was not appropriate for individuals with an ID due to their cognitive deficits. However, this notion has been challenged, and there is growing recognition that individuals with a cognitive impairment, including an ID may benefit from psychological treatments, such as cognitive behavioural therapy (CBT; Taylor et al. 2008). Indeed, the clinical guidelines published in the UK recommend CBT for individuals with milder ID, to

treat depression or subclinical depressive symptoms (National Institute for Health and Care Excellence 2016).

There is a wealth of research on the effectiveness of CBT for anxiety disorders in the general population which include social anxiety disorder, specific phobia, generalised anxiety disorder (GAD), obsessive–compulsive disorder (OCD) and post-traumatic stress disorder (PTSD). CBT may be conceptualised as a class of interventions that are collaborative, structured and skills-orientated, which focus on the interaction between thoughts, feelings and behaviours. It involves attempting to modify unhelpful thoughts, behaviours or both to improve emotional responses (Kaczkurkin & Foa 2015). The treatment components of CBT for anxiety may vary between interventions but can include psychoeducation, relaxation, cognitive strategies, exposure, social skills training and problem solving (Hofmann et al. 2012).

There is a growing body of evidence that has evaluated the effectiveness of CBT in managing problemswith anxiety, anger, and depression in the ID population (Vereenooghe & Langdon 2013; Osugo & Cooper 2016). A meta-analysis of psychological therapies for children and adults with ID found CBT to be at least moderately effective in treating anger and depression with emerging evidence for anxiety (Vereenooghe & Langdon 2013). A scoping review found equivocal evidence for the effectiveness of CBT in improving anxiety for individuals with ID (Unwin et al. 2016). However, amongst these studies, few utilised standardised criteria related to a diagnosis or specific level of anxiety symptomatology and no studies were controlled studies. Nonetheless, qualitative data have shown that CBT is generally perceived as feasible and was well-tolerated amongst clients and carers (Unwin et al. 2016). A subsequent systematic review reported positive outcomes of CBT interventions on anxiety for people with ID (Dagnan et al. 2018); however, the majority of the reviewed literature consisted of single case studies. In addition, some studies included in the systematic review included participants with mixed presentations in a transdiagnostic group, whereby the effects of CBT may have been diluted due to the combination of anxious and depressed participants in treatment outcomes. Furthermore, none of the reviews have included children with an ID up until this point.

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© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

Therefore, the existing literature investigating the effectiveness of CBT on anxiety in people with ID is ambiguous, but positive results are emerging.

There are some challenges associated with delivering CBT to the ID population. Individuals with ID may present with significant deficits in attention, learning, memory, executive function and language, all cognitive abilities required for effective CBT (Hronis et al. 2017). Hronis et al. (2017) contend that while it may be challenging to adapt CBT interventions to an individual’s unique cognitive profile, it should not preclude their participation in CBT. Moreover, there may be greater difficulties adapting CBT for certain presentations of anxiety for those with ID. For example, cognitive interventions may be more central to interventions targeting conditions such as PTSD and GAD, in comparison with specific phobia (Arch & Craske 2009) and therefore may be more difficult to adapt. Some pertinent recommendations include simplification of techniques, adjusting language, utilising activities, using directive methods, using flexible methods and involving caregivers (e.g. assign homework or rehearsals at home with the help of support persons). For children with ID, modifications include shorter sessions, engaging in implicit learning processes (e.g. role play), using visual aids and presenting information numerous times (Hronis et al. 2017).

With increased interest in anxiety interventions for the ID population over recent years and with growing insights into its potential utility and effectiveness for the ID population, a critical review of the literature on CBT treatment for anxiety in the ID group is warranted. This is pertinent considering the growing literature in the field since the previous systematic review was conducted by Dagnan et al. (2018); therefore, this study seeks to update the field of knowledge. Furthermore, this study will attempt to provide a more focused exploration of the evidence base compared with previous reviews, by applying more stringent exclusion criteria whereby only studies with interventions targeting anxiety disorders (rather than transdiagnostic groups) will be included; it is also the first systematic review to examine the effectiveness of CBT for children with anxiety and ID.

This systematic review aims to encourage awareness of the current literature around whether CBT is appropriate and effective for individuals with ID and an anxiety disorder. Moreover, it aims to

identify which adaptations to CBT and which components of CBT are currently being utilised for individuals with ID, as well as a critical analysis of the strengths and limitations and the clinical challenges within this field of research.

Materials and methods

The reporting of this systematic review was guided by the standards of the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA; Moher et al. 2009; Page et al. 2021) statement. Methods of the analysis and inclusion criteria were specified in advance and documented in a protocol (registration number: CRD42020208353).

Eligibility criteria

Inclusion and exclusion criteria

Table 1 demonstrates the inclusion and exclusion criteria of this systematic review. Published studies were required to meet the following criteria: (1) to evaluate the effectiveness of CBT interventions for individuals with ID and anxiety; (2) to include participants who (a) were formally diagnosed with ID using current or older editions of any internationally recognised diagnostic criteria (e.g. Diagnostic and Statistical Manual or the International Classification of Diseases), (b) were found to demonstrate an IQ below 70 through formal cognitive testing, (c) provided previous reports from appropriate health practitioners confirming the diagnosis of ID or (d) had confirmed involvement with a community ID service provider; (3) participants were required to demonstrate anxiety through (a) a formal diagnosis using current or older editions of any recognised diagnostic criteria for anxiety, (b) report elevated levels of anxiety symptoms using a validated measure anxiety (including non-specified anxiety) or (c) demonstrate elevated anxiety that was observed through a clinical assessment or interview with a qualified health practitioner; (4) to report at least two assessment time points (e.g. baseline and post- intervention); and (5) to have been published in a peer review journal in the English language.

The current systematic review excluded studies that included a non-anxious disorder as the primary target of intervention (e.g. a mood disorder). If there was evidence that the participants experienced anxiety

818 Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

as their primary concern, comorbidities such as ASD and mood disorder such as major depressive disorder were included. Studies were excluded if the intervention did not include a specific cognitive component or if there was another non-CBT-based psychological therapy present in parallel to the intervention, whereby it would be impossible to tease apart the specific efficacy of CBT treatment on anxiety symptomology. Dissertations, book chapters, expert reviews and conference abstracts without the full text, and protocols without data were excluded. Restrictions were not placed on the age of participants in the studies included.

ID has replaced older terms, such as ‘mental retardation’, and is recognised alongside other terms such as ‘learning disability’ in the UK, and therefore, participants meeting these diagnoses were considered. Anxiety disorders that would be considered included the following: GAD; specific phobia; panic disorder; agoraphobia; social anxiety disorder; selective mutism; separation anxiety

disorder; and anxiety disorder that was induced by a substance or medication or that was due to another medical condition. OCD, acute stress disorder and PTSD were also included, as these disorders were previously considered to be anxiety disorders in previous editions of recognised diagnostic criteria.

The systematic review was limited to studies investigating CBT interventions and included studies evaluating CBT cognitive and behavioural techniques, such as cognitive restructuring; psychoeducation; behavioural activation; social skills training; relaxation; or coping skills (Cuijpers et al. 2008). In line with Dagnan et al. (2018), interventions that were considered to be a part of the wider family of CBT, such as behavioural activation or third-wave variants of CBT (e.g. mindfulness and acceptance commitment therapy), were only included if there was a specific cognitive component.

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Table 1 The inclusion and exclusion criteria for the current systematic review

Inclusion criteria Exclusion criteria

Study topic Evaluated the effectiveness of CBT for individuals with ID and anxiety

The primary target of the intervention was a non-anxious disorder (e.g. mood disorder)

Participant Evidence of an ID through:

• Formal diagnostic assessment • Cognitive testing (IQ < 70) • Previous reports from health practitioners or, • Involvement with a community ID provider • Includes evidence for ‘mental retardation’

or a ‘learning disability’ in UK Evidence of anxiety through:

• Formal diagnostic assessment of anxiety • Elevated anxiety on a validated measure • Interview/assessment with a qualified

health practitioner Comorbidities (e.g. ASD and mood disorders) were included if anxiety was identified as the primary concern All ages

Study type/design At least two assessment time points (e.g. baseline and post-treatment)

Dissertations, book chapters, expert reviews and conference abstracts without the full text and protocolsPublished in a peer review journal in English

Intervention CBT intervention with at least a cognitive component A non-CBT based psychological therapy was also present

ASD, autism spectrum disorder; CBT, cognitive behavioural therapy; ID, intellectual disability.

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

Information sources

The following databases were accessed: CINAHL (1937 to August 2021), EMBASE (1974 to August 2021), Medline (1946 to August 2021), PsycINFO (1806 to August 2021), Psychology and Behavioural Sciences Collection (1965 to August 2021) and Scopus (1875 to August 2021). For each database, no date limit was applied. The last search was run on 10

August 2021 by two independent researchers. Subject headings and keywords were used where available.

Search strategy

The search strategy was adapted for each specific data base. The search terms were as follows: ((learn* or intellect*) adj2 (disorder* or impair* or disab* or dysfunction*) OR mental retardation OR mental* handicap*) AND (anxi* OR phobi* OR obsessive compulsive disorder OR panic disorder OR agoraphobia OR mutism OR GAD OR OCD OR PTSDOR ((post traumatic stress or post traumatic or overanxi* or avoidan* or internali* or panic*) adj2 disorder*)) AND (cognitive behavi* therapy OR cbt OR mindfulness OR acceptance and commitment therapy OR dialectical behavi* therapy OR ACT OR MBCT OR DBT).

Study selection

In accordance with the PRISMA guidance to reporting (Moher et al. 2009; Page et al. 2021), the process for selecting studies commenced by removing duplicate articles. This was achieved using the online EndNote programme, which can automatically remove duplicates. Further duplicates were removed manually. The remaining results were screened on the basis of the title, the abstract, the participant section and other selection and exclusion criteria. The reference lists were then screened. Two independent researchers reviewed these papers in an unblinded, standardised manner. Disagreements between reviewers were resolved by a third independent reviewer.

Data collection process and synthesis of results

One researcher extracted information from each included study, utilising a pre-designed data extraction form. Information extracted from the selected studies included: aims of the study, country

of origin, participant characteristics, recruitment, intervention characteristics, medication use, study characteristics and measures, results, and quality appraisal. The results were presented and synthesised in the tables. Table 2 presents the results of pre–post intervention studies whereas Table 3 presents the results of the case studies and series. The degree of the effect sizes was interpreted utilising Ferguson’s recommendations for social science data (2009), whereby the effect sizes for r ranged from minimal (.20), moderate (.50) and strong (.80) and the effect sizes for ωp

2 ranged from minimal (.04), moderate (.25) and strong (.64). Where necessary and possible the mean and standard deviations of the outcome measures for anxiety severity were calculated.

In addition, information was collected by one researcher in relation to the treatment components of the included studies. The treatment descriptions and/or treatment manuals were examined, and the following characteristics were recorded: psychoeducation (providing didactic instruction on the nature of anxiety, information on the connection between thoughts, feelings and behaviours); relaxation strategies (diaphragmatic breathing, progressive muscle relaxation); cognitive strategies (identifying and categorising distorted automatic thoughts, cognitive restructuring, Socratic questioning, downward arrow technique, behavioural experiments); and exposure techniques (in vivo or imaginal). In addition, strategies were further specified to examine the presence of cognitive restructuring (cognitive challenging) and thought replacement, also known as self-instructional training (Willner 2005), which involves reminding oneself of coping statements. When coding the specific treatment components of each intervention, the author’s description of the purpose of the treatment component was carefully considered.

Methodological quality

The methodological quality of each study was appraised using established quality assessment tools by the National Institutes of Health (NIH). The NIH has developed a set of quality assessment tools that are tailored to various study designs, such as before-and- after (pre–post) studies with no control group (National Institutes of Health 2014a) and case series designs (National Institutes of Health 2014b). The

820 Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

821

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be ha vi ou

rs .

• Pa rt ic ip an t 2 :1 3 se ss io ns

ov er

1 0 m on

th s.

• C on

du ct ed

at th e

pa rt ic ip an t’s

ho m e

• A D IS -IV

,c lin ic ia n

ad m in is te re d

se m i-s tr uc tu re d in te rv ie w

co nd uc te d w ith

pa re nt s,

de sig ne d to

as se ss

D SM

-IV m oo

d di so rd er s.

• C BC

L, pa re nt

re po

rt of

be ha vi ou

ra l

an d em

ot io na lp

ro bl em

s. •

R C M A S, m ea su re

of an xi et y

ad ap te d fo r pa re nt s.

• M A SC

,m ea su re

of an xi et y,

ad ap te d fo r pa re nt s.

O ne

pa rt ic ip an t co m pl et ed

a se lf- re po

rt as

w el l.

• W

ee kl y ra tin

gs of

an xi et y

by pa re nt

an d ch ild .

• BA

I-Y ,s el f-r ep or t m ea su re

of an xi et y in

yo ut h.

(B la ke le y- Sm

ith et

al .

20 21 )

U SA

• N = 2 3 (6

fe m al es ),

1 2 – 1 9 ye ar s ol d

• 4 pa rt ic ip an ts dr op

pe d ou

t •

ID an d A SD

(d id

no t

sp ec ify

th e le ve ls ).

• Pr e- Po

st st ud y

• Sy m pt om

s of

so ci al an xi et y,

sp ec ifi c ph ob

ia ,g en er al is ed

an xi et y,

se pa ra tio

n an xi et y an d ob

se ss iv e–

co m pu ls iv e fe ar s.

• Fa m ily -fo

cu se d

C BT

in te rv en tio

n w hi ch

w as

ad ap te d

fr om

an ex is tin

g ev id en ce -b as ed

m an ua l,

Fa ci ng

Y ou

r Fe ar s

(R ea ve n et

al .2

0 1 1 )

• G ro up

in te rv en tio

n w ith

2 – 4 ad ol es ce nt s

an d th ei r pa re nt s

• 1 4 × 4 5 – 6 0 m in

se ss io ns

ov er

1 4 w ee ks

• D id

no t re po

rt th e tr ai ni ng

of th e fa ci lit at or s

• A D A M S, a sc re en er

fo r

ps yc hi at ri c di so rd er s

• SC

A R ED

-P ar en t V er si on

, m ea su re s an xi et y

• FS SC

-R w hi ch

m ea su re s

av oi da nc e to w ar ds

fe ar ed

si tu at io ns

• A BC

-C w hi ch

m ea su re s th e

se ve ri ty

of pr ob

le m

be ha vi ou

rs

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

822

T ab

le 2.

(C on tin

ue d)

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

Ph ill ip s & K le in -T as m an

(2 00 9)

U SA

• R es ul ts fr om

th e A D IS re ve al ed

th at

th e

pa rt ic ip an t no

lo ng er

m et

cr ite

ri a fo r

G A D ,O

C D ,h

ow ev er

sh e st ill m et

th e

cr ite

ri a fo r Sp ec ifi c Ph ob

ia .

• W

ee kl y ra tin

g an d qu es tio

nn ai re

m ea su re s di d no

t de m on

st ra te

an im pr ov em

en t

in an xi et y.

• M ix ed

re su lts

fo r pa rt ic ip an t tw

o- so m e

im pr ov em

en ts in

m ea su re s.

• PT

as se ss m en t co nd uc te d

6 m on

th s an d 2 m on

th s

af te r th e co nc lu si on

of th e in te rv en tio

n fo r ea ch

pa rt ic ip an t.

1. C le ar

st ud y qu es tio

n 2.

C le ar

ca se

de fi ni tio

n 3.

C on

se cu tiv e ca se s

4. C om

pa ra bl e su bj ec ts

5. C le ar

in te rv en tio

n 6.

O M

de fi ne d,

va lid

& re lia bl e

7. A de qu at e fo llo w -u p le ng th

8. St at is tic al m et ho

ds w el l-d

es cr ib ed

9. R es ul ts

w el l-d

es cr ib ed

Q ua lit y ra tin

g:

Y Y N R

Y Y Y Y N /A

Y G oo

d (B la ke le y- Sm

ith et

al .

20 21 )

U SA

• Si g re du ct io n in

an xi et y sc or es

w as

ob se rv ed

at PT

co m pa re d w ith

BL on

al lm

ea su re s [A D A M S (ω

2 p = .4 5 ),

SC A R ED

-P (ω

2 p = .1 4 ) an d FS SC

-R (n

= 1 6 ;ω

2 p = .2 0 )] .

• Si g re du ct io n in

so ci al av oi da nc e

(ω 2 p = .5 0 ), ob

se ss iv e–

co m pu ls iv e

sy m pt om

s (ω

2 p = .1 8 ), de pr es si on

(ω 2 p = .2 3 ), an d m oo

d sy m pt om

s (ω

2 p = .2 0 ) w er e ob

se rv ed

on th e

A D A M S sc al e fr om

BL to

PT .

• A si g re du ct io n in

le th ar gy

w as

ob se rv ed

(ω 2 p = .1 4 ), on

th e

A BC

fr om

BL to

PT .

• A nx ie ty

m ea su re

re lie d on

ly on

pa re nt -r ep or t

• A nx ie ty

di so rd er s w er e no

t fo rm

al ly di ag no

se d

• D id

no t re po

rt on

th e le ve lo

ft ra in in g

of th e fa ci lit at or s

• N o fo llo w -u p ou

tc om

e da ta

• O ne

m ea su re

w as

no t ad m in is te re d to

al lp

ar tic ip an ts (F SS C -R )

1. C le ar

ob je ct iv es

2. C le ar

el ig ib ili ty

cr ite

ri a

3. R ep re se nt at iv e pa rt ic ip an ts

4. A ll el ig ib le pe rs on

s en ro lle d

5. Su ffi ci en t sa m pl e si ze

6. In te rv en tio

n 7.

O M

de fi ne d,

va lid ,r el ia bl e

8. A ss es so r bl in di ng

9. Fo

llo w -u p 20 % or

le ss ?

10 .S ta tis tic al an al ys is

11 .M

ul tip

le O M

12 .I nd iv id ua l-l ev el ou

tc om

e fo r

gr ou

p- le ve li nt en tio

ns Q ua lit y ra tin

g:

Y Y Y Y N R

Y Y N Y Y N N /A

Fa ir

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

quality assessment tool for case series designs was also applied to case reports, as there were limited tools available for this study design and because the tool captured important aspects to assess which are relevant to case studies. These tools were selected as they assessed the internal validity and risk of bias in a similar manner. The quality assessments were undertaken by two independent researchers. Any disagreements (N = 16, and 15%) were resolved through discussions with a third party (co-author, M. P.) and were re-coded by the independent third party (co-author, M. P.). These tools informed the reviewers’ critical appraisal of the studies and their classification of the studies as ‘good’, ‘fair’ or ‘poor’. Results of the overall quality assessments are reported in Tables 2 and 3.

Results

Study selection

Figure 1 presents the flowchart describing the inclusion and exclusion of studies.

Study characteristics

Five of the nine included studies were uncontrolled before and after (pre–post) studies, two were case studies and two were case series. Tables 2–6 show the characteristics of studies across various groups of participants.

Participants

The selected studies included a total of 60 participants, aged between 12 and 73 years. The majority of studies included individuals with a mild ID (Marwood & Hewitt 2013; Stuart et al. 2014; Carrigan & Allez 2017; Roberts & Kwan 2018; Giannaki & Hewitt 2021). Four studies included participants with borderline or moderate ID (Lindsay et al. 1997; Douglass et al. 2007; Phillips & Klein- Tasman 2009; Roberts & Kwan 2018). One study did not specify the participants’ level of ID but reported a range of intellectual functioning for participants (IQ = 40–79; Blakeley-Smith et al. 2021). ID was confirmed by a written report from a qualified clinician (Roberts & Kwan 2018), by formally testing IQ and adaptive functioning (Lindsay et al. 1997; Phillips & Klein-Tasman 2009; Stuart et al. 2014;

823

†

T he

ou tc om

e m ea su re s pr ov id e ba se lin e an d po

st -t re at m en t da ta .

‡

T he

m et ho

do lo gi ca lq ua lit y of

st ud ie s w as

as se ss ed

us in g th e N IH

Q ua lit y A ss es sm

en tT

oo lf or

Be fo re -A fte

r (P re -P os t) St ud ie s W

ith N o C on

tr ol

G ro up .1 .C

le ar

ob je ct iv es

= W

as th e st ud y qu es tio

n or

ob je ct iv e

cl ea rl y st at ed ?; 2.

C le ar

el ig ib ili ty

cr ite

ri a = W

er e el ig ib ili ty /s el ec tio

n cr ite

ri a fo r th e st ud y po

pu la tio

n pr es pe ci fi ed

an d cl ea rl y de sc ri be d? ;3

.R ep re se nt at iv e pa rt ic ip an ts

= W

er e th e pa rt ic ip an ts

in th e st ud y re p-

re se nt at iv e of

th os e w ho

w ou

ld be

el ig ib le fo r th e te st /s er vi ce /in te rv en tio

n in th e ge ne ra lo

r cl in ic al po

pu la tio

n of

in te re st ?; 4. A ll el ig ib le pe rs on

s en ro lle d = W

er e al le lig ib le pa rt ic ip an ts th at

m et

th e pr es pe ci fi ed

en tr y cr ite

ri a en ro lle d? ;5

.S uf fi ci en t sa m pl e si ze

= W

as th e sa m pl e si ze

su ffi ci en tly

la rg e to

pr ov id e co nfi de nc e in

th e fi nd in gs ?; 6.

In te rv en tio

n = W

as th e te st /s er vi ce /in te rv en tio

n cl ea rl y de sc ri be d an d de liv er ed

co ns is te nt ly ac ro ss

th e st ud y po

pu la tio

n? ;7

= O M

de fi ne d,

va lid ,r el ia bl e = W

er e th e ou

tc om

e m ea su re s pr es pe ci fi ed ,c le ar ly de fi ne d,

va lid ,r el ia bl e, an d as se ss ed

co ns is te nt ly ac ro ss

al ls tu dy

pa rt ic ip an ts ?; 8.

A ss es so r bl in di ng

= W

er e th e pe op

le as se ss in g th e ou

tc om

es bl in de d to

th e pa rt ic ip an ts ’e xp os ur es /in te rv en tio

ns ?; 9. Fo

llo w -u p 20 % or

le ss ?=

W as

th e lo ss to

fo llo w -u p af te r ba se lin e 20 % or

le ss ?W

er e th os e lo st

to fo llo w -u p ac co un te d fo r in th e an al ys is ?; 10 .S ta tis tic al an al ys is = D id th e st at is tic al m et ho

ds ex am

in e ch an ge s in ou

tc om

e m ea su re s fr om

be fo re

to af te r th e in te rv en tio

n? W

er e st at is tic al te st s do

ne th at pr ov id ed

p va lu es

fo r th e pr e- to -p os t ch an ge s? ;1

1. M ul tip

le O M

= W

er e ou

tc om

e m ea su re s of

in te re st

ta ke n m ul tip

le tim

es be fo re

th e in te rv en tio

n an d m ul tip

le tim

es af te r th e in te rv en tio

n (…

)? ;1

2. In di vi du al -le

ve l

ou tc om

e fo r gr ou

p- le ve li nt en tio

n = If th e in te rv en tio

n w as

co nd uc te d at

a gr ou

p le ve l( …

) di d th e st at is tic al an al ys is ta ke

in to

ac co un t th e us e of

in di vi du al -le

ve ld

at a to

de te rm

in e ef fe ct s at

th e gr ou

p le ve l?

N ot e: A BC

-C ,A

be rr an t Be

ha vi ou

r C he ck lis t- C om

m un ity

(A m an

et al .1

99 5) ;A

D A M S, A nx ie ty

D ep re ss io n an d M oo

d Sc al e (E sb en se n et

al .2

00 3) ;A

D IS -IV

,A nx ie ty

D is or de rs

In te rv ie w

Sc he du le fo r D SM

-IV ,

pa re nt

ve rs io n (S ilv er m an

& A lb an o 19 96

); BA

I-Y ,B

ec k A nx ie ty

In ve nt or y Y ou

th (B ec k et

al .2

00 1) ;C

BC L,

C hi ld

Be ha vi ou

r C he ck lis t (A ch en ba ch

& R es co rl a 20 01

); FS SC

-R ,F ea r Su rv ey

Sc he du le fo r

C hi ld re n- R ev is ed

(O lle nd ic k 19 83

); M A SC

,M ul tid

im en si on

al A nx ie ty

Sc al e fo r C hi ld re n (M

ar ch

19 97

); R C M A S, R ev is ed

C hi ld re n’ s M an ife st A nx ie ty

Sc al e (R ey no

ld s & R ic hm

on d 19 98

); SC

A R E- P, Sc re en

fo r C hi ld

A nx ie ty

R el at ed

D is or de rs ,P

ar en t V er si on

(B ir m ah er

et al .1

99 9) .

A D H D ,a tt en tio

n- de fi ci t/ hy pe ra ct iv ity

di so rd er ;B

L, ba se lin e; C BT

,c og ni tiv e be ha vi ou

ra lt he ra py ;C

D ,c an no

t de te rm

in e; M ,g ro up

m ea n; N ,n o;

N ,n um

be r of

pa rt ic ip an ts ;N

/A ,n ot

ap pl ic ab le ;N

R ,n ot

re po

rt ed ;

O D D ,o

pp os iti on

al de fi an t di so rd er ;O

M ,o

ut co m e m ea su re ;P

T ,p

os t- tr ea tm

en t; SD

,s ta nd ar d de vi at io n;

si g, si gn ifi ca nt ;s ig ,s ig ni fi ca nt ;Y

,y es .

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

824

T ab

le 3

S ys te m at ic

re vi ew

su m m ar y of

fi n d in gs

fo r C B T

fo r ad

u lt s w it h ge n er al is ed

an xi et y or

m ix ed

an xi et y (n

= 4 )

R ef er en

ce an

d co

un tr y

St ud

y/ sa m pl in g ch

ar ac

te ri st ic s

A nx

ie ty

co nd

it io ns

In te rv en

ti on

de si gn

an d de

ta ils

M ea

su re s us ed

D ou

gl as s et

al .( 20 07 )

U K

• N = 6 (4

fe m al es ), 2 2 – 6 5 ye ar s ol d

• 1 pa rt ic ip an t dr op

pe d ou

t be fo re

th e be gi nn in g of

th e gr ou

p •

Bo rd er lin e- m od

er at e ID

• Pr e- po

st st ud y

• G en er al is ed

an xi et y

sy m pt om

s •

Pr e & po

st in te rv en tio

n no

co nt ro l,

m ix ed

m et ho

d de sig n

• C BT

w ith

th e as sis ta nc e

of a su pp or t pe rs on

• 1 2 × 1 2 0 m in

se ss io ns

ov er

1 2 w ee ks

• G ro up

th er ap y, fa ce -t o- fa ce

in th e co m m un ity

• C lin ic ia ns :A

ss is ta nt

Ps yc ho

lo gi st ,

T ra in ee

C lin ic al

Ps yc ho

lo gi st ,L

ea rn in g

D is ab ili ty

N ur se

an d

O cc up at io na lT

he ra pi st T ec hn ic ia n

• G A S- ID ,a

se lf- re po

rt m ea su re

of an xi et y

(G ia nn ak i&

H ew

itt 20 21 )

U K

• N = 4 (3

fe m al es ), ag ed

2 1 – 5 5 ye ar s of

ag e

• M ild

ID •

Pa rt ic ip an ts w er e

re fe rr ed

to th e ps yc ho

lo gy

se rv ic e fo r pe op

le w ith

a le ar ni ng

di sa bi lit y

• Pr e- po

st st ud y

• Sy m pt om

s re la te d

to ge ne ra lis ed

an xi et y an d

sp ec ifi c ph ob

ia

• Pr e & po

st in te rv en tio

n no

co nt ro l

• Fo

llo w -u p at

4 m on

th s

• C BT

w ith

th e as sis ta nc e

of a su pp or t pe rs on

• C BT

co nc ep ts w er e

ba se d on

Be ck

(1 9 7 9 )

• 7 w ee kl y se ss io ns

(d id

no t re po

rt on

th e

du ra tio

n of

se ss io ns )

• G ro up

th er ap y, fa ce -t o- fa ce

in th e co m m un ity

• C lin ic ia ns :t ra in ee

cl in ic al ps yc ho

lo gi st s

an d as si st an t ps yc ho

lo gi st s

• G A S- ID

se lf- re po

rt •

H oN

O S- LD

,a se lf- re po

rt m ea su re

of gl ob

al fu nc tio

ni ng .

• PT

O S- ID ,s el f-

an d su pp or te r-

re po

rt m ea su re

of ps yc ho

lo gi ca ld

is tr es s

an d w el lb ei ng .

M ar w oo

d an d H ew

itt (2 01 3)

U K

• N = 8 (4

fe m al es ), ag ed

1 7 – 7 3 ye ar s of

ag e

• M ild

ID •

Pa rt ic ip an ts w er e re cr ui te d

th ro ug h N H S

re fe rr al s to

th e ps yc ho

lo gi ca l

se rv ic e fo r

pe op

le w ith

a le ar ni ng

di sa bi lit y

• In cl ud ed

pa rt ic ip an ts w ith

au tis m

an d de pr es si on

. •

Pr e- po

st st ud y

• In cl ud ed

sy m pt om

s of

sp ec ifi c ph ob

ia ,

so ci al an xi et y,

ge ne ra lis ed

an xi et y,

an d ob

se ss io na la nx ie ty .

• Pr e & po

st in te rv en tio

n no

co nt ro l,

m ix ed

m et ho

d de sig n

• C BT

th er ap y w ith

th e as si st an ce

of a su pp or t pe rs on

• 6 × 6 0 m in

se ss io ns

ov er

6 w ee ks

• G ro up

th er ap y, fa ce -t o- fa ce

in th e co m m un ity

• C lin ic ia ns :t ra in ee

ps yc ho

lo gi st an d

an as si st an t ps yc ho

lo gi st

• G A S- ID

se lf- re po

rt •

Q oL

• H oN

O S– LD

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

825

T ab

le 3.

(C on tin

ue d)

R ef er en

ce an

d co

un tr y

St ud

y/ sa m pl in g ch

ar ac

te ri st ic s

A nx

ie ty

co nd

it io ns

In te rv en

ti on

de si gn

an d de

ta ils

M ea

su re s us ed

R ob

er ts & K w an

(2 01 8)

A U S

• N = 1 3 (7

fe m al es ), ag ed

1 8 – 5 5 ye ar s of

ag e

• M ild

(n = 1 0 ) an d m od

er at e

(n = 3 ) ID

• Pa rt ic ip an ts w er e re cr ui te d

th ro ug h ad ve rt is em

en ts

to se rv ic es

an d gr ou

ps fo r

pe op

le w ith

ID (e .g .r es pi te

se rv ic es ,

m en ta lh

ea lth

se rv ic es )

• Pr e- po

st st ud y

• D ia gn os es

of sp ec ifi c

ph ob

ia ,s oc ia l

ph ob

ia an d

ge ne ra lis ed

an xi et y di so rd er

• Pr e & po

st in te rv en tio

n no

co nt ro l

• C BT

gr ou

p th er ap y w ith

op tio

na l

as si st an ce

of a th er ap y pa rt ne r

• 6 × 9 0 m in

se ss io ns

ov er

6 w ee ks

• G ro up

th er ap y, fa ce -t o- fa ce

• T he

th er ap y m at er ia ls

w er e cr ea te d by

a re gi st er ed

cl in ic al ps yc ho

lo gi st ,

th e fi rs t au th or

an d w er e ba se d

on C BT

pr og ra m m es

fo r

ty pi ca lly

de ve lo pi ng

in di vi du al s w ith

an xi et y

• G A S- ID

co m pl et ed

by th e

pa rt ic ip an ts an d th ei r ca re r

or su pp or t w or ke r

• PA

S- A D D ,a

cl in ic al in te rv ie w ,

w as

ad m in ist er ed

by a cl in ic al ps yc ho

lo gi st to

di ag no

se an xi et y di so rd er s

• M in iP

A S- A D D

w as

ad m in ist er ed

by a cl in ic al nu rs e

co ns ul ta nt

to sc re en

fo r

an xi et y di so rd er s

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

D ou

gl as s et

al .( 20 07 )

U K

• H al ft he

pa rt ic ip an ts re po

rt ed

im pr ov em

en ts

in an xi et y sc or es

at PT

• Si g lo w er

se lf- re po

rt ed

an xi et y (G

A S- ID )

or 2 pa rt ic ip an ts

• Sl ig ht

in cr ea se s in

se lf- re po

rt ed

an xi et y

(G A S- ID ) fo r 3 pa rt ic ip an ts

• A ll pa rt ic ip an ts re po

rt ed

le ar ni ng

ne w

w ay s

of co pi ng

w ith

th ei r an xi et y at

PT •

N o at tr iti on

• G ro up

m ea n of

an xi et y

re du ce d fr om

BL (M

= 2 5 .5 0 ,S D = 3 .0 4 ) to

PT (M

= 2 1 .8 3 ,S D = 7 .3 8 )§

• D id

no t ad eq ua te ly de sc ri be

th e in te rv en tio

n •

D id

no t ve ri fy th e ID

of al lp

ar tic ip an ts ,l ev el s

of ID

w er e de te rm

in ed

by cl in ic al ju dg em

en t

• A nx ie ty

di so rd er

w as

no t

fo rm

al ly di ag no

se d

• N o fo llo w -u p da ta

• La ck

of m ul tip

le -b as el in es

1. C le ar

ob je ct iv es

2. C le ar

el ig ib ili ty

cr ite

ri a

3. R ep re se nt at iv e pa rt ic ip an ts

4. A ll el ig ib le pe rs on

s en ro lle d

5. Su ffi ci en t sa m pl e si ze

6. In te rv en tio

n 7.

O M

de fi ne d,

va lid ,r el ia bl e

8. A ss es so r bl in di ng

9. Fo

llo w -u p 20 % or

le ss ?

10 .S ta tis tic al an al ys is

11 .M

ul tip

le O M

12 .I nd iv id ua l-l ev el ou

tc om

e fo r gr ou

p- le ve li nt en tio

ns Q ua lit y ra tin

g:

Y Y Y Y N N Y N Y N N N /A

Po or

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

826

T ab

le 3.

(C on tin

ue d)

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

(G ia nn ak i&

H ew

itt 20 21 )

U K

• A ll fo ur

pa rt ic ip an ts ex pe ri en ce d

si g re du ct io ns

in an xi et y fr om

BL to

PT (P

< .0 5 );

th es e ga in s re m ai ne d la rg el y

at fo llo w -u p.

• T he

gr ou

p m ea n fo r

an xi et y re du ce d fr om

BL (M

= 2 5 .3 ,S D = 5 .9 0 ) to

PT (M

= 1 3 ,S D = 3 .8 2 ).

• T he

ov er al ll ev el of

di st re ss

w as

re du ce d w he re as

th e ov er al ll ev el of

gl ob

al fu nc tio

ni ng

in th e gr ou

p w as

en ha nc ed

in th e gr ou

p at

PT co m pa re d w ith

BL .

• N o at tr iti on

.

• Sm

al ls am

pl e si ze ,n

o co nt ro l

gr ou

p, th er eb y re du ci ng

th e

re lia bi lit y an d ge ne ra lis ab ili ty

of th e st ud y

• N o fo rm

al di ag no

si s of

an xi et y

re qu ir ed

fo r pa rt ic ip at io n

• La ck

of co ns is te nc y in

th e

pr oc ed ur es

(e .g .o

ne pa rt ic ip an t

jo in ed

th e gr ou

p in

th e fo ur th

w ee k)

1. C le ar

ob je ct iv es

2. C le ar

el ig ib ili ty

cr ite

ri a

3. R ep re se nt at iv e pa rt ic ip an ts

4. A ll el ig ib le pe rs on

s en ro lle d

5. Su ffi ci en t sa m pl e si ze

6. In te rv en tio

n 7.

O M

de fi ne d,

va lid ,r el ia bl e

8. A ss es so r bl in di ng

9. Fo

llo w -u p 20 % or

le ss ?

10 .S ta tis tic al an al ys is

11 .M

ul tip

le O M

12 .I nd iv id ua l-l ev el ou

tc om

e fo r gr ou

p- le ve li nt en tio

ns Q ua lit y ra tin

g:

Y Y Y N R

N C D

Y N Y Y N N /A

Fa ir

M ar w oo

d an d H ew

itt (2 01 3)

U K

• Si g re du ct io n in

se lf- re po

rt ed

an xi et y

(G A S- ID ) fo r 2 pa rt ic ip an ts .

• Im

pr ov em

en ts in

th e gr ou

p m ea ns

in an xi et y fr om

BL (M

= 2 6 .8 8 ,S D = 7 .2 5 ) to

PT (M

= 2 3 .3 8 ,S D = 7 .4 2 )§ .

• Im

pr ov em

en ts in

gr ou

p m ea ns

in gl ob

al fu nc tio

ni ng

an d qu al ity

of lif e w er e

ob se rv ed

fr om

BL to

PT .

• So m e pa rt ic ip an ts

ha d co m or bi di tie

s (a ut is m

sp ec tr um

di so rd er

an d de pr es si on

) •

C lin ic al cu t- of fs fo r an xi et y w er e no

t pr ed et er m in ed

fo r el ig ib ili ty

• Sm

al ls am

pl e si ze ,

po te nt ia lly

lim iti ng

th e

ge ne ra lis ab ili ty

of th e st ud y

• N o fo llo w -u p da ta

1. C le ar

ob je ct iv es

2. C le ar

el ig ib ili ty

cr ite

ri a

3. R ep re se nt at iv e pa rt ic ip an ts

4. A ll el ig ib le pe rs on

s en ro lle d

5. Su ffi ci en t sa m pl e si ze

6. In te rv en tio

n 7.

O M

de fi ne d,

va lid ,r el ia bl e

8. A ss es so r bl in di ng

9. Fo

llo w -u p 20 % or

le ss ?

10 .S ta tis tic al an al ys is

11 .M

ul tip

le O M

12 .I nd iv id ua l-l ev el ou

tc om

e fo r gr ou

p- le ve li nt en tio

ns Q ua lit y ra tin

g:

Y N Y C D

N Y Y N Y N N N /A

Po or

R ob

er ts & K w an

(2 01 8)

A U S

• Si g re du ct io n in

th e pr op

or tio

n of

pa rt ic ip an ts di ag no

se d

as cl in ic al ly an xi ou

s fr om

BL to

PT (7 6 .9 % – 7 .7 % ).

• N o fo llo w -u p ou

tc om

e da ta

• A si ng le cl in ic al ad m in ist er ed

th e

cl in ic al as se ss m en ts at

BL an d PT

w ho

w as

no t

bl in de d to

th e co nd iti on

s

1. C le ar

ob je ct iv es

2. C le ar

el ig ib ili ty

cr ite

ri a

3. R ep re se nt at iv e pa rt ic ip an ts

4. A ll el ig ib le pe rs on

s en ro lle d

5. Su ffi ci en t sa m pl e si ze

Y Y Y N R

N

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

827

T ab

le 3.

(C on tin

ue d)

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

• 1 1 pa rt ic ip an ts no

lo ng er

m et

cr ite

ri a fo r th ei r pr im ar y

an xi et y di ag no

si s at

PT .

• Si g re du ct io ns

in an xi et y

(G A S- ID ) w er e fo un d

fo r se lf- re po

rt (z

= �3

.0 6 ,

P <

.0 1 ) an d

in fo rm

an t- re po

rt (z

= �2

.4 9 ,

P = .0 1 ) fo llo w in g

th e in te rv en tio

n. Bo

th th e

ef fe ct

si ze s w er e m od

er at e

(r = .6 0 fo r se lf- ra tin

g an d r = .4 9

fo r in fo rm

an t ra tin

gs ).

• La ck

of m ul tip

le ba se lin es

an d a co nt ro lg ro up

6. In te rv en tio

n 7.

O M

de fi ne d,

va lid ,r el ia bl e

8. A ss es so r bl in di ng

9. Fo

llo w -u p 20 % or

le ss ?

10 .S ta tis tic al an al ys is

11 .M

ul tip

le O M

12 .I nd iv id ua l-l ev el ou

tc om

e fo r gr ou

p- le ve li nt en tio

ns Q ua lit y ra tin

g:

Y Y N N R

Y N N /A

Fa ir

†

T he

ou tc om

e m ea su re s pr ov id e ba se lin e an d po

st -t re at m en t da ta .

‡

T he

m et ho

do lo gi ca lq

ua lit y of

st ud ie s w as

as se ss ed

us in g th e N IH

Q ua lit y A ss es sm

en t T oo

lf or

Be fo re -A fte

r (P re -P os t) St ud ie s W

ith N o C on

tr ol

G ro up .1

.C le ar

ob je ct iv es

= 1.

W as

th e st ud y qu es tio

n or

ob je ct iv e cl ea rl y st at ed ?; 2.

C le ar

el ig ib ili ty

cr ite

ri a = W

er e el ig ib ili ty /s el ec tio

n cr ite

ri a fo r th e st ud y po

pu la tio

n pr es pe ci fi ed

an d cl ea rl y de sc ri be d? ;3

.R ep re se nt at iv e pa rt ic ip an ts

= W

er e th e pa rt ic ip an ts

in th e

st ud y re pr es en ta tiv e of

th os e w ho

w ou

ld be

el ig ib le fo r th e te st /s er vi ce /in te rv en tio

n in

th e ge ne ra lo

r cl in ic al po

pu la tio

n of

in te re st ?; 4.

A ll el ig ib le pe rs on

s en ro lle d = W

er e al le lig ib le pa rt ic ip an ts

th at

m et

th e

pr es pe ci fi ed

en tr y cr ite

ri a en ro lle d? ;5 .S uf fi ci en ts am

pl e si ze

= W

as th e sa m pl e si ze

su ffi ci en tly

la rg e to

pr ov id e co nfi de nc e in th e fi nd in gs ?; 6. In te rv en tio

n = W

as th e te st /s er vi ce /in te rv en tio

n cl ea rl y de sc ri be d an d

de liv er ed

co ns is te nt ly ac ro ss th e st ud y po

pu la tio

n? ;7

= O M

de fi ne d, va lid ,r el ia bl e = W

er e th e ou

tc om

e m ea su re s pr es pe ci fi ed ,c le ar ly de fi ne d, va lid ,r el ia bl e, an d as se ss ed

co ns is te nt ly ac ro ss al ls tu dy

pa rt ic ip an ts ?;

8. A ss es so r bl in di ng

= W

er e th e pe op

le as se ss in g th e ou

tc om

es bl in de d to

th e pa rt ic ip an ts ’e xp os ur es /in te rv en tio

ns ?; 9. Fo

llo w -u p 20 % or

le ss ?=

W as

th e lo ss to

fo llo w -u p af te r ba se lin e 20 % or

le ss ?W

er e th os e

lo st to

fo llo w -u p ac co un te d fo r in

th e an al ys is ?; 10 .S ta tis tic al an al ys is = D id th e st at is tic al m et ho

ds ex am

in e ch an ge s in

ou tc om

e m ea su re s fr om

be fo re

to af te r th e in te rv en tio

n? W

er e st at is tic al te st s do

ne th at

pr ov id ed

p va lu es

fo r th e pr e- to -p os t ch an ge s? ;1

1. M ul tip

le O M

= W

er e ou

tc om

e m ea su re s of

in te re st

ta ke n m ul tip

le tim

es be fo re

th e in te rv en tio

n an d m ul tip

le tim

es af te r th e in te rv en tio

n (…

)? ;1

2. In di vi du al -le

ve lo

ut co m e fo r gr ou

p- le ve li nt en tio

n = If th e in te rv en tio

n w as

co nd uc te d at

a gr ou

p le ve l( … ) di d th e st at is tic al an al ys is ta ke

in to

ac co un t th e us e of

in di vi du al -le

ve ld

at a to

de te rm

in e ef fe ct s at

th e

gr ou

p le ve l?

§ A n es tim

at e w as

ca lc ul at ed

fo r th e gr ou

p m ea n an d st an da rd

de vi at io n.

N ot e: G A S- ID ,G

la sg ow

A nx ie ty

Sc al e fo r pe op

le w ith

an In te lle ct ua lD

is ab ili ty (M

in dh am

& Es pi e 20 03

); H oN

O S- LD

,H ea lth

of th e N at io n Br ie fO

ut co m e M ea su re

fo r Pe op

le w ith

Le ar ni ng

D is ab ili tie

s an d M en ta l

H ea lth

N ee ds

(R oy

et al .2

00 2) ;P

A S- A D D ,P

sy ch ia tr ic A ss es sm

en t Sc he du le

fo r A du lts

w ith

D ev el op

m en ta lD

is ab ili tie

s (M

os s & Fr ie dl an de r 20 11

); Q oL

,Q ua lit y of

Li fe

Sc al e (A nd re w s & W

ith ey

19 76

). A U S, A us tr al ia ;B

L, ba se lin e; C BT

,c og ni tiv e be ha vi ou

ra lt he ra py ;C

D ,c an no

td et er m in e; M ,g ro up

m ea n; N ,n o; N ,n um

be r of

pa rt ic ip an ts ;N

/A ,n ot

ap pl ic ab le ;N

R ,n ot

re po

rt ed ;O

M ,o ut co m e m ea su re ;P T ,p os t-

tr ea tm

en t; SD

,s ta nd ar d de vi at io n;

si g, si gn ifi ca nt ;s ig ,s ig ni fi ca nt ;U

K ,U

ni te d K in gd om

;Y ,y es .

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

Blakeley-Smith et al. 2021), or through health records (Marwood & Hewitt 2013). Douglass et al. (2007) reported that the ID of clients was largely determined based on the clinical judgement of the researchers. Giannaki and Hewitt (2021) and Carrigan and Allez (2017) did not clarify how the participants’ level

of ID was determined or verified; however, these participants had confirmed involvement with a community ID service provider. Overall, the majority of participants were recruited by convenience or were referred by professionals to a private service.

828

FIGURE 1. PRISMA 2009 flow diagram highlights the exclusion of articles at each search stage

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

829

T ab

le 4

S ys te m at ic

re vi ew

su m m ar y of

fi n d in gs

fo r C B T

fo r ad

u lt s w it h P T S D

sy m pt om

s (n

= 1 )

R ef er en

ce an

d co

un tr y

St ud

y/ sa m pl in g ch

ar ac

te ri st ic s

A nx

ie ty

co nd

it io ns

In te rv en

ti on

de si gn

an d de

ta ils

M ea

su re s us ed

C ar ri ga n an d A lle z (2 01 7)

U K

• N = 1 m al e, 2 6 ye ar s ol d

• M ild

ID an d A SD

• C as e st ud y

• PT

SD sy m pt om

s •

C as e st ud y

• T ra um

a- fo cu se d C BT

w ith

el em

en ts of

co gn iti ve

th er ap y

ba se d on

th e ap pr oa ch

by (E hl er s et

al .2

0 0 5 )

• 1 2 × 6 0 m in

se ss io ns

ov er

1 2 w ee ks

• In di vi du al se ss io ns ,f ac e- to

fa ce

in th e co m m un ity

• C lin ic ia n w as

a cl in ic al ps yc ho

lo gi st

• C R IE S- 8 w hi ch

is a

se lf- re po

rt m ea su re

of in tr us io ns

an d av oi da nc e

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

C ar ri ga n an d A lle z (2 01 7)

U K

• T he

C R IE S- 8 sc or e

re du ce d fr om

3 2 to

1 1 ,

w hi ch

w as

be lo w

th e

cl in ic al cu t of fo

f1 7 (Y ul e 1 9 9 2 ).

• Pa rt ic ip an t an d hi s

pa re nt

re po

rt ed

im pr ov em

en ts in

PT SD

sy m pt om

s.

• N o fo rm

al di ag no

si s of

ID ,

un cl ea r w he th er

do cu m en ta tio

n w as

si gh te d to

co nfi rm

di ag no

si s

• A br ie fs cr ee ni ng

to ol

de ve lo pe d fo r th e ge ne ra l

po pu la tio

n w as

us ed

to as se ss

PT SD

sy m pt om

s, an d th er ef or e m ay

be su bj ec t

to m ea su re m en t er ro r

• La ck

of m ul tip

le ba se lin es

• N o fo llo w -u p da ta

1. C le ar

st ud y qu es tio

n 2.

C le ar

ca se

de fi ni tio

n 3.

C on

se cu tiv e ca se s

4. C om

pa ra bl e su bj ec ts

5. C le ar

in te rv en tio

n 6.

O M

de fi ne d,

va lid

& re lia bl e

7. A de qu at e fo llo w -u p le ng th

8. St at is tic al m et ho

ds w el l-d

es cr ib ed

9. R es ul ts w el l-d

es cr ib ed

Q ua lit y ra tin

g:

Y Y N /A

N /A Y N Y N /A Y

Po or

†

T he

ou tc om

e m ea su re s pr ov id e ba se lin e an d po

st -t re at m en t da ta .

‡

T he

m et ho

do lo gi ca lq

ua lit y of

st ud ie s w as

as se ss ed

us in g th e N IH

Q ua lit y A ss es sm

en t of

C as e Se ri es

St ud ie s. 1.

C le ar

st ud y qu es tio

n = W

as th e st ud y qu es tio

n or

ob je ct iv e cl ea rl y st at ed ?; 2.

C le ar

ca se

de fi ni tio

n = W

as th e st ud y po

pu la tio

n cl ea rl y an d fu lly

de sc ri be d, in cl ud in g a ca se

de fi ni tio

n? ;3 .C

on se cu tiv e ca se s = W

er e th e ca se s co ns ec ut iv e? ;4 .C

om pa ra bl e su bj ec ts = W

er e th e su bj ec ts co m pa ra bl e? ;5 .C

le ar

in te rv en tio

n = W

as th e in te rv en tio

n cl ea rl y de sc ri be d? ;6 .O

M de fi ne d, va lid

& re lia bl e = W

er e th e ou

tc om

e m ea su re s cl ea rl y de fi ne d, va lid ,r el ia bl e, an d im pl em

en te d co ns is te nt ly ac ro ss

al ls tu dy

pa rt ic ip an ts ?; 7.

A de qu at e fo llo w -u p le ng th

= W

as th e le ng th

of fo llo w -u p ad eq ua te ?; 8.

St at is tic al m et ho

ds w el l-d

es cr ib ed

= W

er e th e st at is tic al m et ho

ds w el l-d

es cr ib ed ?; 9.

R es ul ts

w el l-d

es cr ib ed

= W

er e th e re su lts

w el l-

de sc ri be d?

N ot e: C R IE S- 8,

R ev is ed

C hi ld

Im pa ct

of Ev en ts

Sc al e (S m ith

et al .2

00 3) .

BL ,b as el in e; C BT

,c og ni tiv e be ha vi ou

ra lt he ra py ;C

D ,c an no

td et er m in e; M ,g ro up

m ea n; m in ,m

in ut es ;N

,n o;

N ,n um

be r of

pa rt ic ip an ts ;N

/A ,n ot

ap pl ic ab le ;N

R ,n ot

re po

rt ed ;O

M ,o

ut co m e m ea su re ;P T ,p os t-

tr ea tm

en t; SD

,s ta nd ar d de vi at io n;

si g, si gn ifi ca nt ;s ig ,s ig ni fi ca nt ;U

K ,U

ni te d K in gd om

;Y ,y es .

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

830

T ab

le 5

S ys te m at ic

re vi ew

su m m ar y of

fi n d in gs

fo r C B T

fo r ad

u lt s w it h n on

-s pe

ci fi ed

an xi et y (n

= 2 )

R ef er en

ce an

d co

un tr y

St ud

y/ sa m pl in g ch

ar ac

te ri st ic s

A nx

ie ty

co nd

it io ns

In te rv en

ti on

de si gn

an d de

ta ils

M ea

su re s us ed

Li nd sa y et

al .( 19 97 )

U K

• N = 2 ,1 9 an d 2 7 ye ar -o ld

m al es

• M ild

in te lle ct ua ld

is ab ili ty

(IQ = 6 6 – 6 7 )

• El ev at ed

le ve ls of

de pr es si on

fo r 1 pa rt ic ip an t

• N on

-s pe ci fi ed

an xi et y

(e le va te d le ve ls of

an xi et y)

• C as e se ri es

• In di vi du al co gn iti ve

fo r an xi et y

fo r ad ul ts ,b

as ed

up on

Be ck

et al .’s

(1 9 7 9 ) m an ua l

• Pa rt ic ip an t 1 :4

1 in di vi du al

se ss io ns

ov er

1 -y ea r ta rg et ed

an xi et y

• Pa rt ic ip an t 2 :1 4 se ss io ns

ov er

1 4 w ee ks

• Fo

llo w -u p at

3 – an d 6 -m

on th s PT

fo r ea ch

pa rt ic ip an t re sp ec tiv el y

• BA

I, a se lf- re po

rt m ea su re

of an xi et y.

• BD

I, a se lf- re po

rt m ea su re

of de pr es si on

.

St ua rt

et al .( 20 14 )

U K

• N = 1 fe m al e, 4 0 ye ar s ol d

• M ild

ID •

A nx io us

an d de pr es si ve

sy m pt om

s •

Pa rt ic ip an t w as

re fe rr ed

fr om

th ei r G P to

th e C LD

T

• N on

-s pe ci fi ed

an xi et y

• C as e st ud y

• Be

ha vi ou

ra lly

fo cu se d in di vi du al C BT

• 7 se ss io ns

ov er

1 2 w ee ks

(t he

du ra tio

n of

se ss io ns

w as

no t re po

rt ed )

• G A S- ID

se lf- re po

rt m ea su re

an xi et y

• G D S- LD

,s el f-r ep or t

m ea su re

of de pr es si on

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

Li nd sa y et

al .( 19 97 )

U K

• Si g re du ct io ns

in an xi et y fo r bo

th pa rt ic ip an ts ;t he ir BA

Is co re

re du ce d

fr om

ve ry

hi gh

le ve ls at

BL to

to le ra bl e

or no

rm al le ve ls at

PT .G

ai ns

w er e

m ai nt ai ne d at

fo llo w -u p (3 – 6 m on

th s

af te r PT

). •

A si g re du ct io n in

an xi et y- re la te d

co gn iti on

s w as

ob se rv ed .

• A si g im pr ov em

en t in

de pr es si on

se ve ri ty

w as

al so

ob se rv ed

fo r on

e pa rt ic ip an t.

• N o fo rm

al di ag no

si s of

an xi et y

• D id

no t re po

rt on

th e le ve lo

fI D

• D id

no t re po

rt on

th e le ve lo

f tr ai ni ng

of th e fa ci lit at or s

1. C le ar

st ud y qu es tio

n 2.

C le ar

ca se

de fi ni tio

n 3.

C on

se cu tiv e ca se s

4. C om

pa ra bl e su bj ec ts

5. C le ar

in te rv en tio

n 6.

O M

de fi ne d,

va lid

& re lia bl e

7. A de qu at e fo llo w -u p le ng th

8. St at is tic al m et ho

ds w el l-d

es cr ib ed

9. R es ul ts

w el l-d

es cr ib ed

Q ua lit y ra tin

g:

Y N N R Y Y Y Y N /A Y

G oo

d St ua rt

et al .( 20 14 )

U K

• T he

an xi et y se ve ri ty

sc or e re du ce d fr om

BL to

PT ;h

ow ev er ,i t w as

st ill ab ov e

th e cl in ic al cu t- of fp

oi nt

at PT

. •

T he

de pr es si on

se ve ri ty

sc or e re du ce d

fr om

BL to

PT ;h

ow ev er ,i t w as

st ill

ab ov e th e cl in ic al cu t- of fp

oi nt

at PT

.

• In su ffi ci en t de ta il re ga rd in g th e

tr ea tm

en t co m po

ne nt s w as

pr ov id ed

• T he re

w as

no fo llo w -u p ou

tc om

e da ta

• La ck

of m ul tip

le ba se lin e

• N o fo rm

al di ag no

si s of

an xi et y

1. C le ar

st ud y qu es tio

n 2.

C le ar

ca se

de fi ni tio

n 3.

C on

se cu tiv e ca se s

4. C om

pa ra bl e su bj ec ts

5. C le ar

in te rv en tio

n 6.

O M

de fi ne d,

va lid

& re lia bl e

7. A de qu at e fo llo w -u p le ng th

Y Y N /A

N /A

Y Y N

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

Elevated anxiety symptoms were largely determined using predetermined clinical cut-offs on standardised measures of anxiety symptomatology. One study reported that eligible participants demonstrated ‘anxiety issues’ in need of management, but did not elaborate on how this was determined (Marwood & Hewitt 2013). Two studies utilised a clinical diagnostic interview to determine anxiety diagnoses (Phillips & Klein-Tasman 2009; Roberts & Kwan 2018). Participants presented with a variety of anxious symptoms that were often associated with generalised anxiety, specific phobia and social anxiety.

Methodological quality

Tables 2–5 demonstrate the study strengths and weaknesses of the included studies, based on the NIH quality assessment tools. As can be seen in Tables 2–5, overall, two studies were classified as ‘good’, three studies were rated as ‘fair’ and four studies were rated as ‘poor’ on the overall quality ratings. Methodological strengths of the reviewed studies included the following: low attrition rates [80% (n = 4 out of 5) of pre–post studies]; representative samples [100% (n = 5 out of 5)]; clear study objectives [100% (n = 9 out of 9) of all studies]; and clear selection criteria for participants [80% (n = 4 out of 5)]. Sixty per cent of studies (n = 3 out of 5) included appropriate statistical analyses. Weaknesses of the reviewed studies included the following: lack of multiple baseline [0% (n = 5 out of 5)] and lack of blinded assessors [0% (n = 5 out of 5)].

Cognitive behavioural therapy delivery

Table 6 summarises the main features of each of the reviewed studies, such as therapy format (individual or group therapy), participant characteristics (e.g. age and ID levels), CBT treatment components utilised and types of modifications made. The reviewed studies used CBT interventions that ranged from 6 to 41 sessions in total, with a duration of 1–2 h per session. As can be seen from Table 6, four studies utilised individual CBT sessions, and the remaining five adopted a group-based CBT delivery. The group interventions included compulsory or optional support persons in therapy. All interventions were facilitated by a therapist. The qualifications of the therapist varied across studies and included registered

831

T ab

le 5.

(C on tin

ue d)

R ef er en

ce an

d co

un tr y

M ai n fi nd

in gs

† L im

it at io ns

M et ho

do lo gi ca

lq ua

lit y‡

8. St at is tic al m et ho

ds w el l-d

es cr ib ed

9. R es ul ts

w el l-d

es cr ib ed

Q ua lit y ra tin

g:

N /A

Y Po or

†

T he

ou tc om

e m ea su re s pr ov id e ba se lin e an d po

st -t re at m en t da ta .

‡

T he

m et ho

do lo gi ca lq

ua lit y of

st ud ie s w as

as se ss ed

us in g th e N IH

Q ua lit y A ss es sm

en t of

C as e Se ri es

St ud ie s. 1.

C le ar

st ud y qu es tio

n = W

as th e st ud y qu es tio

n or

ob je ct iv e cl ea rl y st at ed ?; 2.

C le ar

ca se

de fi ni tio

n = W

as th e st ud y po

pu la tio

n cl ea rl y an d fu lly

de sc ri be d,

in cl ud in g a ca se

de fi ni tio

n? ;3

.C on

se cu tiv e ca se s = W

er e th e ca se s co ns ec ut iv e? ;4

.C om

pa ra bl e su bj ec ts

= W

er e th e su bj ec ts

co m pa ra bl e? ;5

. C le ar

in te rv en tio

n = W

as th e in te rv en tio

n cl ea rl y de sc ri be d? ;6

.O M

de fi ne d,

va lid

& re lia bl e = W

er e th e ou

tc om

e m ea su re s cl ea rl y de fi ne d,

va lid ,r el ia bl e, an d im pl em

en te d co ns is te nt ly ac ro ss

al ls tu dy

pa rt ic –

ip an ts ?; 7. A de qu at e fo llo w -u p le ng th

= W

as th e le ng th

of fo llo w -u p ad eq ua te ?; 8. St at is tic al m et ho

ds w el l-d

es cr ib ed

= W

er e th e st at is tic al m et ho

ds w el l-d

es cr ib ed ?; 9. R es ul ts w el l-d

es cr ib ed

= W

er e th e re su lts

w el l-d

es cr ib ed ?

N ot e: BA

I, Be

ck A nx ie ty

In ve nt or y (B ec k et

al .1

98 8) ;B

D I, Be

ck D ep re ss io n In ve nt or y; G A S- ID ,G

la sg ow

A nx ie ty

Sc al e fo r pe op

le w ith

an In te lle ct ua lD

is ab ili ty

(M in dh am

& Es pi e 20 03

); G D S- LD

,G la sg ow

D ep re ss io n Sc al e fo r Pe op

le w ith

a Le ar ni ng

D is ab ili ty

(C ut hi ll et

al .2

00 3) .

BL ,b as el in e; C BT

,c og ni tiv e be ha vi ou

ra lt he ra py ;C

D ,c an no

td et er m in e; M ,g ro up

m ea n; m in ,m

in ut es ;N

,n o;

N ,n um

be r of

pa rt ic ip an ts ;N

/A ,n ot

ap pl ic ab le ;N

R ,n ot

re po

rt ed ;O

M ,o

ut co m e m ea su re ;P T ,p os t-

tr ea tm

en t; SD

,s ta nd ar d de vi at io n;

si g, si gn ifi ca nt ;s ig ,s ig ni fi ca nt ;U

K ,U

ni te d K in gd om

;Y ,y es .

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

832

T ab

le 6

S ys te m at ic

re vi ew

su m m ar y of

fi n d in gs

ac ro ss

st u d ie s (N

= 9 )

T he

ra py

P ar ti ci pa

nt s

A nx

ie ty

ty pe

T re at m en

t co

m po

ne nt s

St ud

y In di vi du

al th er ap

y G ro

up th er ap

y B or

de rl in e/

m ild

ID M od

er at e/

se ve

re ID

N on

-s pe

ci fi ed

ID le ve

l U nd

er 18

ye ar s

A nx

ie ty

no t sp ec

ifi ed

G en

er al is ed

an xi et y

Sp ec

ifi c

ph ob

ia So

ci al

an xi et y

P T SD

Se pa

ra ti on

an xi et y

O bs es si ve

co m pu

ls iv e

P sy ch

o ed

uc at io n

R el ax

at io n

E xp

o su re

(B la ke le y- Sm

ith et

al .2

02 1)

• •

• •

• •

• •

• •

Ph ill ip s &

K le in -T as m an

(2 00 9)

• •

• •†

•† •

•† •

• •

•

D ou

gl as s

et al .( 20 07

) •

• •

• •

• •

(G ia nn ak i

& H ew

itt 20 21 )

• •

• •

• •

M ar w oo

d an d

H ew

itt (2 01 3)

• •

• •

• •

• •

R ob

er ts

& K w an

(2 01 8)

• •

• •†

•† •†

• •

•

C ar ri ga n an d

A lle z (2 01 7)

• •

• •

Li nd sa y et

al .( 19 97

) •

• •

•

St ua rt

et al .( 20 14

) •

• •

• •

T re at m en

t co

m po

ne nt s

M od

ifi ca

ti on

s

St ud

y D is tr ac

ti on

B eh

av io ur

al ac

ti va

ti on

Id en

ti fy in g

em ot io ns

Id en

ti fy

un he

lp fu l

th ou

gh ts

C og

ni ti ve

re st ru

ct ur

in g

T ho

ug ht

re pl ac

em en

t B eh

av io ur

al ex

pe ri m en

ts Im

ag in al

re liv

in g

C op

in g

st ra te gi es

H om

ew or

k ta sk s

R eh

ea rs in g

R ol ep

la ys

Su pp

o rt

pe rs on

in vo

lv ed

A do

pt ed

sm al le r

gr o up

s M o de

lli ng

V is ua

lA id s

S lo w er

pa ce

A da

pt la ng

ua ge

/ si m pl ify

(B la ke le y- Sm

ith et

al .2

02 1)

• •

• •

• •

• •

• •

Ph ill ip s &

K le in -T as m an

(2 00 9)

• •

• •

• •

• •

•

D ou

gl as s

et al .( 20 07

) •

• •

• •

• •

• •

• •

(G ia nn ak i

& H ew

itt 20 21 )

• •

• •

• •

• •

• •

M ar w oo

d an d

H ew

itt (2 01 3)

• •

• •

• •

• •

R ob

er ts

& K w an

(2 01 8)

• •

• •

• •

• •

• •

C ar ri ga n an d

A lle z (2 01 7)

• •

• •

•

Li nd sa y et

al .( 19 97

) •

• •

• •

• •

St ua rt

et al .( 20 14

) •

• •

• •

†

Pa rt ic ip an ts

m et

th e cr ite

ri a fo r th e an xi et y di so rd er

lis te d (o th er

st ud ie s in cl ud ed

pa rt ic ip an ts

w ith

su bc lin ic al di ag no

se s or

fe ar s re la te d to

th os e do

m ai ns ).

A D H D ,a tt en tio

n- de fi ci t/ hy pe ra ct iv ity

di so rd er ;I D ,i nt el le ct ua ld

is ab ili ty .

Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

clinical psychologists (Carrigan & Allez 2017), assistant or trainee psychologists (Douglass et al. 2007; Marwood & Hewitt 2013; Stuart et al. 2014; Giannaki & Hewitt 2021), learning disability nurses and occupational therapists (Douglass et al. 2007). Four studies did not specify the qualifications or training of the therapists (Lindsay et al. 1997; Phillips & Klein-Tasman 2009; Roberts & Kwan 2018; Blakeley-Smith et al. 2021).

Three studies reported that participants continued taking prescribed medication for anxiety or comorbid low mood throughout the study; however, the type and dose remained consistent for a considerable time prior and during the intervention (Marwood & Hewitt 2013; Stuart et al. 2014; Blakeley-Smith et al. 2021). The remainder of the studies did not report on medication use.

Child studies

Mixed anxiety

Amongst studies with child participants, one included participants aged 12–19 years (Phillips & Klein- Tasman 2009; Blakeley-Smith et al. 2021) and the other 13 years (Phillips & Klein-Tasman 2009), with various anxiety disorders and fears (e.g. generalised anxiety, separation anxiety and specific phobia) (refer to Table 2). Blakeley-Smith et al. (2021) included children/adolescents with a comorbid diagnosis of ASD and Phillips and Klein-Tasman (2009) included adolescents with comorbid diagnoses of Williams syndrome, ADHD and oppositional defiant disorder (ODD; Phillips & Klein-Tasman 2009).

Treatment components and adaptations. Several components were included across the two studies to treat various anxiety disorders; these included psychoeducation, relaxation, exposure, identifying unhelpful thoughts, thought replacement, cognitive restructuring and strategies to promote emotion regulation (refer to Table 6). However, Phillips and Klein-Tasman (2009) reported that exposure and cognitive restructuring were not routinely used or focused on.

Adaptations that were commonly applied across the two studies to accommodate for the needs of the children/adolescents included adopting a slower pace, rehearsing, involving parents in the intervention, and tailored support for problem behaviours.

Blakeley-Smith et al. (2021) reported that they adapted the intervention based off stakeholder input gained through informal focus groups, parent interviews and treatment acceptability ratings. Various modifications to the programme included reducing the length of sessions, having smaller groups, greater parental involvement in sessions, and providing information sessions to parents on various topics (e.g. the difficulty detecting anxiety in individuals with ID and ASD, and the interplay between anxiety and problem behaviour). They also reported including visual supports to teach strategies, video modelling, hands on activities and reinforcement of brave behaviour. On the other hand, Phillips and Klein-Tasman (2009) reported including techniques to encourage participants to cope with teasing to assist them with their anxiety in social situations and strategies to manage oppositional behaviours and emotional outbursts.

Strategies were tailored to the children’s/adolescents’ cognitive abilities. Blakeley-Smith et al. adapted the delivery of strategies based on the participants’ expressive language ability. For example, participants with communication difficulties were provided helpful thoughts (e.g. ‘I can do this, I’m brave’) with a visual structure instead of delivering cognitive restructuring (‘fight fear with facts’). Similarly, Phillips and Klein-Tasman (2009) delivered more straightforward behavioural strategies, such as thought replacement (‘adaptive self- statements’) instead of cognitive restructuring when participants had difficulties comprehending the material.

Treatment outcomes. Across the child/adolescent studies, there were positive results on the effectiveness of CBT on anxiety. Blakeley-Smith et al. (2021) found that following the intervention, there were significant improvements in the group mean of anxiety and minimal and moderate effect sizes on parent-reported measures of anxiety [e.g. Anxiety Depression and Mood Scale (ωp

2 = .45) and the total FSSC-R (ωp

2 = .20)]. Moreover, there were significant reductions in the total score and separation anxiety subscale of the SCARED-P, but not in the other subscales; they proposed that this may be because some domains of anxiety (e.g. generalised or social anxiety) are more difficult for parents to observe, relying more heavily on a child’s verbalisation of

833 Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

worry. Phillips and Klein-Tasman (2009) also found improvements in anxiety severity across some measures whereas others indicated no change or an exacerbation of symptoms. Following the intervention, the two participants no longer met the diagnostic criteria for various anxiety disorders recognised at pre-treatment; however, some remained clinical (Phillips & Klein-Tasman 2009).

Additional outcomes. Parent acceptability ratings, attendance and completion rates indicated that Blakeley-Smith et al.’s (2021) intervention was feasible to implement and valued by families. They also reported a significant reduction in symptoms of depression (ωp

2 = .23), mood symptoms (ωp 2 = .20)

and lethargy (ωp 2 = .14) in the group following a CBT

intervention, compared with baseline; still, these effects sizes were only minimal. However, significant reductions in symptoms of irritability were not observed, suggesting that problem behaviours were not impacted by the intervention. Phillips and Klein- Tasman (2009) also found significant improvements in emotional–behavioural difficulties, where one participant no longer met the criteria for ODD following treatment; however, it should be noted a considerable portion of the intervention focused on strategies specifically addressing emotional outbursts.

Adult studies

Generalised anxiety or mixed anxiety

Three studies included adults with diagnoses or symptoms of generalised anxiety, specific phobia, social anxiety or OCD (Marwood & Hewitt 2013; Roberts & Kwan 2018; Giannaki & Hewitt 2021) and one study included adults experiencing generalised anxiety (Douglass et al. 2007; refer to Table 3). Marwood and Hewitt (2013) included adult participants with comorbid diagnoses of ASD and depression.

Treatment component and adaptations. Various treatment components were included across the adult studies with generalised and mixed anxiety, such as psychoeducation and relaxation. Other behavioural strategies, such as distraction and thought replacement, were reported less (Douglass et al. 2007; Marwood & Hewitt 2013). While identifying unhelpful thoughts was a common component, only

one study included cognitive restructuring, whereby unhelpful thoughts were actively challenged (Roberts & Kwan 2018). Moreover, only two studies (Douglass et al. 2007; Roberts & Kwan 2018) incorporated exposure, providing minimal detail on this component; it was, however, unclear whether Douglass et al. (2007) only delivered this component to one participant.

Various modifications to the interventions for anxiety were referenced across the adult studies. Some studies modified treatments based off recommendations in the literature for adults with ID (e.g. Whitehouse et al. 2006) or the clinician’s previous work with the population (Roberts & Kwan 2018). Common adaptations applied across studies involved incorporating visual supports, roleplays, rehearsing strategies, simplifying language and including a support person. Studies proposed that including a support person who knew the participant well, was motivated and consistent was beneficial to treatment outcomes (Douglass et al. 2007; Marwood & Hewitt 2013). Other modifications that were used less included using games and stories to teach materials, having shorter sessions, adopting a slower pace and using concrete examples (Douglass et al. 2007; Giannaki & Hewitt 2021).

Across studies cognitive components were modified to cater for a participant’s cognitive capacity. Roberts and Kwan (2018) incorporated activities, roleplays and metaphors (e.g. changing red to green traffic light thoughts) to adapt cognitive challenging to meet the needs of adults with mild ID. Individuals with moderate ID benefited from additional modifications, such as extensive modelling, rehearsing and prompting. Douglass et al. (2007) endorsed a flexible approach when delivering cognitive strategies. When participants had difficulties identifying negative thoughts, roleplays were offered (which was of limited benefit) and participants were encouraged to practise ‘generic coping statements’.

Treatment outcomes. Across the adult studies examining the effectiveness of CBT for mixed anxiety presentations, significant reductions in self- and informant-reported anxiety scores were found. Giannaki and Hewitt (2021) found that all four participants in the study demonstrated significant improvements following treatment, which were

834 Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

largely maintained at follow-up. Correspondingly, Roberts and Kwan (2018) found moderate effect sizes for the improvements in self (r = .60) and informant ratings (r = .49) of anxiety severity for participants with mild to moderate ID following treatment. They also found that there was a significant reduction in the proportion of participants diagnosed with an anxiety disorder following the intervention (7.7%; n = 1) compared with that prior to the intervention (7.69%; n = 10).

However, some studies reported mixed results on the effectiveness of CBT on anxiety in adults with ID. For example, Douglass et al. (2007) found a significant reduction in anxiety for two out of six participants who presented with borderline–moderate ID; whereas, slight increases in anxiety were reported for three out of six participants. Similarly, Marwood and Hewitt (2013) observed significant reductions in anxiety for two out of eight participants with mild ID; however, slight increases in anxiety were observed for three participants. Marwood and Hewitt (2013) suggested results may have been impacted by the sensitivity of the measure used.

Additional outcomes. Studies reported improvements across additional measures, such as global functioning, distress, quality of life and mood. Giannaki and Hewitt (2021) observed significant improvements in the overall level of distress for most participants following the intervention, which was largely maintained at follow-up. In addition, significant improvements in global functioning were observed for half of the participants following the intervention. Marwood and Hewitt (2013) also observed improvements in the participants’ quality of life and global functioning following treatment. Furthermore, the completion rates across studies indicated that the interventions were well tolerated (Douglass et al. 2007; Giannaki & Hewitt 2021); however, two studies did not report on attrition rates (Marwood & Hewitt 2013; Roberts & Kwan 2018). Douglass et al. (2007) also found that participants developed coping skills following the intervention and that carers developed a greater understanding of the participants’ difficulties, according to qualitative feedback.

Studies reported on the participants’ ability to learn and demonstrate cognitive strategies. Giannaki and Hewitt (2021) found that during the post-treatment

interviews, no participant demonstrated an understanding of the impact of unhelpful thoughts on anxiety; they proposed this reflected an inability to link thoughts and emotions. Contrastingly, Roberts and Kwan (2018) found that most participants with mild ID were able to demonstrate competence with linking thoughts and emotions and other skills associated with cognitive restructuring (e.g. gather evidence on whether a thought is realistic, generate a more helpful or realistic belief). However, individuals with moderate ID demonstrated limited or no competence across most of these skills (besides identifying unhelpful and helpful thoughts). Therefore, a participant’s proficiency with cognitive restructuring appeared to be impacted by their level of ID.

Post-traumatic stress disorder

Carrigan and Allez (2017) delivered a trauma-focused CBT intervention to a young man with mild ID and ASD and symptoms consistent with PTSD (see Table 4). The intervention incorporated elements of cognitive therapy for PTSD.

Treatment component and adaptations. Carrigan and Allez (2017) delivered a CBT intervention for PTSD, which included psychoeducation on trauma and its sequelae (using a simplified model), cognitive restructuring, behavioural experiments and imaginal reliving. Strategies to address anger outbursts were also introduced towards the beginning of treatment. The authors described making only minimal modifications to cognitive therapy for PTSD. They found that with careful use of Socratic questioning, simplifying the language and explaining metaphors, they were able to help the participant overcome difficulties with applying abstract concepts to his own situation and to see alternate viewpoints when engaging in cognitive restructuring.

Treatment outcomes. There were improvements in the participants’ symptomatology following the intervention, whereby the severity of PTSD symptoms fell within the normal range on a screening measure (Carrigan & Allez 2017). This corresponded with qualitative improvements in the participants’ symptomatology (e.g. improved sleep, reduced

835 Journal of Intellectual Disability Research VOLUME 67 PART 9 SEPTEMBER 2023

G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

nightmares and avoidance) as reported by the participant and their parent.

Non-specified anxiety

Two studies (Lindsay et al. 1997; Stuart et al. 2014) included adult participants with elevated anxiety that was not specified or diagnosed; these participants also presented with low mood, which was not formally assessed (see Table 5).

Treatment components and adaptations. Lindsay et al. (1997) reported delivering cognitive therapy based on the treatment by Beck et al. (1979) whereas Stuart and colleagues described delivering a behaviourally focused CBT intervention. The treatment in both studies included various components which included psychoeducation on the link between thoughts, feelings and behaviours, and noticing or identifying unhelpful thoughts. Lindsay et al.’s (1997) intervention focused on cognitive strategies, consistent with cognitive restructuring; whereas Stuart et al. (2014) focused on behavioural strategies, such as behavioural activation and relaxation.

Lindsay et al. (1997) and Stuart et al. (2014) described adopting various modifications, which included simplifying language, encouraging repetition and having a more activity-based approach. Lindsay et al. (1997) also included adaptions to help participants to elicit thoughts, practise more adaptive ways of thinking and overcome difficulties with abstract thought; these included using role plays, reversing the roles of therapist and client, plotting the sequence of events leading to anxiety and looking at photos of anxiety provoking situations (which may bear some resemblance to exposure practises).

Treatment and additional outcomes. Across the studies treating non-specified anxiety in adults with ID (Lindsay et al. 1997; Stuart et al. 2014), participants demonstrated improvements in the severity of anxiety and depression. Following intervention, Lindsay et al. (1997) found that one participant’s symptoms of anxiety and depression fell within the normal range, whereas Stuart et al. (2014) observed that the participants’ anxiety and low mood remained above the clinical cut-off.

Discussion

This systematic review explored the literature on the effectiveness of CBT for individuals with ID and anxiety. It sought to update the field to include more recent studies and to adopt a more stringent inclusion criteria to focus on studies that target anxiety (rather than transdiagnostic groups), thereby providing a more focused review on anxiety compared with previous reviews. It was also the first systematic review to examine the effectiveness of CBT for children/adolescents with ID and anxiety.

Across the nine studies reviewed, CBT interventions were found to have a positive effect on the severity levels of anxiety in children/adolescents and adults with ID; seven studies reported significant reductions in anxiety for 25%–100% of participants (N = 60) following a CBT intervention. These findings are encouraging; however, in light of the absence of any control groups and other methodological shortcomings, the conclusions that can be drawn are limited. The findings highlight that CBT for children/adolescents and adults with anxiety and mild ID may be feasible and tolerable for a variety of anxiety disorders. Positive outcomes were reported in line with previous reviews on the effectiveness of CBT for adults with ID and anxiety or depression (Unwin et al. 2016; Dagnan et al. 2018). Moreover, improvements generalised to overall functioning, general distress levels, quality of life and mood following the intervention. The results highlight that children/adolescents with various presentations of anxiety and comorbidities can tolerate and benefit from a CBT intervention that is tailored to their cognitive and behavioural needs. It also highlights the importance of a flexible approach in the delivery of strategies.

The studies that showed the most promising results (moderate effect sizes) (Roberts & Kwan 2018; Blakeley-Smith et al. 2021; Giannaki & Hewitt 2021) tended to be the more recent and methodologically rigorous studies, incorporating various cognitive and behavioural components of CBT and more modifications. Interestingly, Blakeley-Smith et al. (2021) and Roberts and Kwan (2018) were the only studies that routinely delivered exposure and cognitive restructuring or thought replacement. In light of the encouraging results, it may indicate the importance of these components in treating anxiety for adults and children with anxiety and ID; this is

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G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

consistent with findings in the TD literature that exposure and cognitive restructuring are key components in treating anxiety (Kaczkurkin & Foa 2015). However, additional case studies that included cognitive techniques (e.g. cognitive restructuring) in the absence of exposure demonstrated positive results indicating emerging evidence for cognitive components for individuals with mild ID (Lindsay et al. 1997; Carrigan & Allez 2017). Across the studies, individuals with mild ID and some with moderate ID appeared to benefit from learning about the link between thoughts, feelings and behaviours, strategies to identify unhelpful thoughts and relaxation strategies to understand and manage anxiety.

Pleasingly, the components of CBT were adapted to the needs of participants with ID across all studies. There was considerable overlap in the adaptations used across studies, regardless of the participants’ age or anxiety disorder. Many of the studies reported utilising adaptations, such as rehearsing techniques, involving a parent/support person, utilising smaller groups and adapting language. These modifications were consistent with the recommendations from the literature (Hronis et al. 2017). Nevertheless, many of the included studies lacked sufficient detail regarding the nature of the modifications, which poses challenges for the replication and development of treatment modifications. Moreover, several studies included modules or strategies in interventions to accommodate comorbidities, problem behaviours and difficulties with emotion regulation; this resulted in significant improvements in behaviours that challenge in some cases (e.g. Phillips & Klein- Tasman 2009). Blakeley-Smith et al. (2021) however proposed that the anxiety intervention was not helpful in targeting problem behaviour and suggested an additional module addressing these concerns may be warranted for some individuals prior to the CBT anxiety intervention. Therefore, future studies may benefit from tailoring intervention to the participants’ cognitive capacity, comorbidities and emotion regulation concerns.

Across the studies, it was apparent that a flexible approach towards the delivery of cognitive skills was beneficial to accommodate for the age and the cognitive capacity of each person. Some children/adolescents and adults with mild ID (or who

were verbally fluent) were found to effectively engage with and implement key components of cognitive restructuring; this included individuals presenting with a variety of anxiety disorders (e.g. generalised anxiety, social anxiety and PTSD). The use of Socratic questioning was found to be important in facilitating the process of challenging negative thoughts and generating more helpful thoughts. Participants with a moderate ID (or emerging language skills) were often able to identify helpful and unhelpful thoughts with certain accommodations but struggled with the use of more conceptual strategies, such as cognitive restructuring. Participants with a moderate ID tended to benefit from a more directive and simplified approach involving the use of thought replacement, whereby participants reminded themselves of coping statements. They also benefited from extensive modelling, rehearsing, prompting and the use of visual aids, regardless of their age. Therefore, cognitive strategies, such as cognitive restructuring, appear to be beneficial and feasible for children and adolescents with mild ID with the use of appropriate modifications and a flexible approach.

Strengths of the studies include using measures that were standardised, valid and reliable, having low attrition rates, and including participants with elevated levels of anxiety. In addition, most of the studies used a measure of anxiety that was developed for the ID population. However, a number of limitations were observed. For example, the studies lacked a control condition and utilised small sample sizes, which underpowered the results. The majority of the studies did not include follow-up assessment to ascertain the long-term maintenance of the intervention, so this remains uncertain. Most of the studies relied upon self-report measures, and no studies included blinded assessors, which increased the risk of bias. Many studies did not utilise appropriate statistical analyses or multiple baseline outcome data, which is important as it allows for inferences to be made about the effectiveness of an intervention. Therefore, despite some strengths, there were considerable methodological limitations across the studies reviewed, so current conclusions are preliminary.

The studies included in this review differed across several domains, which may have affected treatment outcomes. First, there was variability in the inclusion criteria for participants across studies, such that some

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Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

studies required a clinical diagnosis of an anxiety disorder, whereas others required elevated anxiety symptomatology based on questionnaire data. Consequently, some studies may have included participants with subclinical anxiety, resulting in limited reductions in anxiety due to a floor effect. Also, the majority of studies relied upon self-report measures of anxiety, some of which had not been validated in the ID population (e.g. Carrigan & Allez 2017). While self-report measures are desirable in measuring internal states, such as anxiety, the validity of an individual’s self-report may be impacted by factors associated with having an ID; these factors include difficulties with memory, social desirability, response bias and acquiescence (Willner 2005). These challenges in assessment may be alleviated in part by collecting information from multiple informants; however, only two studies included in this review adopted this approach (Phillips & Klein- Tasman 2009; Roberts & Kwan 2018). Therefore, these methodological and assessment limitations may have impacted treatment outcomes.

There was also variability across studies in terms of the delivery of interventions. There was unclear terminology used in interventions across studies. For example, some studies reported including the technique ‘positive self-talk’ (Douglass et al. 2007) while others referred to ‘helpful thoughts’ (Blakeley- Smith et al. 2021), ‘adaptive self-statements’(Phillips & Klein-Tasman 2009) or ‘cognitive challenging’ (Roberts & Kwan 2018). Therefore, the line delineating between specific techniques was at times blurred, especially in cases where there was limited information on the interventions delivered. Finally, there was variability across studies in terms of the skill and training of therapists. Some therapists were trainee psychologists, while others were occupational therapists, endorsed clinical psychologists, or in some cases, it was not reported. Therefore, the outcomes of CBT interventions may have been influenced by the level of training that practitioners received in CBT and in working with individuals with ID. Thus, the studies included have a number of shortcomings and disparities, which may have impacted the treatment findings.

Future research may benefit from addressing these discrepancies and limitations by (1) incorporating more rigorous methodologies, which include a formal diagnosis of anxiety and ID; (2) utilising clear and

universally accepted descriptors of interventions including the treatment components; (3) providing clear details around the procedural modifications made to the delivery of CBT; (4) including follow-up outcome measures and/or multiple outcome data; (5) collecting information from unbiased assessors and/or multiple informants; (6) validating and adapting existing measures of anxiety in the ID population; (7) exploring what modifications to CBT interventions are helpful for individuals with varying levels of ID, particularly through focus groups; (8) utilising large-scale RCTs to evaluate the effectiveness of CBT for the ID population and to explore which CBT components are most effective in treating anxiety and which adaptations may be necessary for varying levels of ID; (9) evaluating more flexible interventions that may be adapted to the verbal competencies of the participants, as seen in the intervention delivered by Blakeley-Smith et al. (2021); and (10) developing evidence-manualised treatments that are tailored to the needs of individuals with mild or moderate ID.

This systematic review employed rigorous criteria to identify eligible studies. Eligible studies were required to present pre- and post-outcome measures of anxiety and did not include interventions, which targeted mixed presentations (i.e. conditions other than anxiety). However, there were several limitations of this systematic review. First, only studies in English were considered, and consequently, most studies were completed in the UK, with some in the USA and Australia. Therefore, the findings of this review may have limited generalisability to other regions or cul- tures. Additionally, the inclusion criteria for the presence of ID allowed for participants who had been referred from an ID service provider; therefore, par- ticipants may have been included that are not truly representative of people with ID. Furthermore, the search terms for ID were narrow and may have re- sulted in some papers examining other related con- ditions (e.g. Fragile X syndrome) being missed. Most of the studies included in this review were composed of participants with a mild ID. Therefore, the findings of the present review may have limited generalisability to participants with moderate or severe ID, and even borderline impaired IQ. Studies were also included that reported on comorbid ASD diagnosis; it is un- clear whether the treatment modifications may gen- eralise to the broader ID population due to the distinct profile of ASD.

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G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the

Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

The current systematic review explored the literature on the effectiveness of CBT for anxiety in individuals with ID, an area that is grossly under- researched. Findings suggest that CBT is a promising treatment for individuals with mild ID and anxiety. Although strong recommendations regarding treatment effectiveness cannot be made at this time due to the limited number of studies and methodological limitations in the research, there is tentative evidence for techniques such as cognitive restructuring and thought replacement and modifications such as visual aids, modelling and smaller groups, at least for those aged 12 years and over. The current review highlights a clear need for future studies to incorporate more scientifically rigorous methodologies to address the limitations identified in this review and identified gaps in the literature. It also reveals that there is a lack of emphasis on the ID population in the mental health literature and that there is a need for more training for practitioners working alongside individuals with ID. Most pertinently, research needs to focus on the development of evidence-based treatment protocols and clinical practise guidelines. Studies on the effectiveness of CBT for different levels of ID with various modifications will better inform the recommendations for treatments for individuals with ID and anxiety. Ultimately, this will assist individuals with ID experiencing anxiety to have access to effective treatments that are tailored to their ability levels and their needs.

Acknowledgements

We extend our gratitude to the reviewers and editors for their feedback.

Open access publishing facilitated by Macquarie University, as part of the Wiley – Macquarie University agreement via the Council of Australian University Librarians.

Source of funding

No external funding was received for the research reported in the paper.

Conflict of interest

There are no conflicts of interest. This is a systematic review and no human ethics approval was required.

Data availability statement

Data available on request.

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Accepted 12 May 2023

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G. Fynn et al. • The effectiveness of cognitive behavioural therapy for individuals with an intellectual disability and anxiety: a systematic review

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Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.

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