Research in Autism Spectrum Disorders 9 (2015) 151–162

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Research in Autism Spectrum Disorders

J ou rna l hom e pa ge : h tt p: / / e e s . e l se v i e r . com / R AS D / de f a ul t . a s p

Cognitive behaviour therapy for adults with autism spectrum disorders

and psychiatric co-morbidity: A review
a,b, c d b,e
Debbie Spain *, Jacqueline Sin , Trudie Chalder , Declan Murphy , Francesca
a
Happe´
a
MRC Social, Genetic and Developmental Psychiatry Centre, Institute of Psychiatry, King’s College London, De Crespigny Park, PO Box 80, London
SE5 8AF, UK
b
Department of Forensic and Neurodevelopmental Sciences, Institute of Psychiatry, King’s College London, De Crespigny Park, PO Box 50, London
SE5 8AF, UK
cFlorence Nightingale School of Nursing & Midwifery, King’s College London, 57 Waterloo Road, London SE1 8WA, UK
d
Department of Psychological Medicine, King’s College London, Weston Education Centre, Cutcombe Road, London SE5 9RJ, UK
e Sackler Centre for Translational Neurodevelopment, Institute of Psychiatry, King’s College London, De Crespigny Park, PO Box 50, London
SE5 8AF, UK

ARTICLE INFO ABSTRACT

Article history: Co-morbid mental health conditions are highly prevalent in autism spectrum disorders (ASD). Cognitive
Received 3 October 2014 Accepted 20 behaviour therapy (CBT) is frequently used to treat these symptoms. Hence, a systematic review was
October 2014 Available online 9 undertaken to synthesise published data about the effectiveness of CBT interventions for adults with ASD
November 2014 and psychiatric co-morbidity. Only six studies met pre-determined review inclusion criteria: two RCTs; one
quasi-experimental study; one case series; and two case studies. Meta-analysis was not possible due to study
Keywords: heterogeneity. A narrative analysis of the data suggested that CBT interventions – including behavioural,
Autism spectrum disorder cognitive, and mindfulness-based techniques – were moderately effective treatments for co-morbid anxiety
Psychiatric co-morbidity and depression symptoms, albeit that sample sizes were small, participant characteristics varied widely, and
Mental health
psychometric properties of self-report outcome measurements utilised in the ASD population remain subject
Cognitive behaviour therapy
to some debate. Several studies described adaptations to standard CBT including an increase in the number
CBT
Mindfulness of sessions, or accommodation of core ASD characteristics and associated neuropsychological impairments
within the therapy process. We suggest further empirical research is needed to

(1) investigate the acceptability and effectiveness of a range of CBT interventions for adults who have ASD
and co-morbidity, and (2) to identify which adaptations are requisite for optimising CBT techniques and
outcomes in this population.
2014 Elsevier Ltd. All rights reserved.

Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 152
1.1. Cognitive behaviour therapy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 152
1.2. Cognitive behaviour therapy for individuals with ASD. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 153
1.3. Rationale for this review . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 153

* Corresponding author at: MRC Social, Genetic and Developmental Psychiatry Centre, Institute of Psychiatry, King’s College London, De Crespigny Park, PO Box 80,
London SE5 8AF, UK. Tel.: +44 207 848 5388; fax: +44 207 848 0650.
E-mail address: debbie.spain@kcl.ac.uk (D. Spain).

http://dx.doi.org/10.1016/j.rasd.2014.10.019
1750-9467/ 2014 Elsevier Ltd. All rights reserved.
 152 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162
2. Methods . . . . . . ..................................................................................... 153

2.1. Search strategy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 153

2.2. Inclusion criteria . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 153
2.3. Exclusion criteria . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 154
2.4. Study selection and data extraction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 154
2.5. Plan for analysis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3. Results ....... ..................................................................................... 155
3.1. Study methods and setting . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3.2. Quality assessment. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3.3. Participant demographics . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3.3.1. Participant clinical diagnoses . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3.3.2. Co-morbid psychiatric diagnosis. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 155
3.4. Structure and content of CBT treatment . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.1. CBT modality . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.2. Manualised treatment protocols . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.3. CBT techniques . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.4. Frequency and duration . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.5. Homework . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 157
3.4.6. Adaptations to CBT . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 158
3.5. Outcomes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 158
3.5.1. Outcome measurement . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 158
3.5.2. Treatment outcomes – effectiveness of CBT. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 158
3.6. Attrition ..................................................................................... 159
3.7. Fidelity to treatment . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 159
4. Discussion . . . . ..................................................................................... 159
4.1. Overview. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 159
4.2. Limitations . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 159
4.3. Clinical practice implications . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 160
4.4. Implications for research . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 160
5. Conclusion . . . . ..................................................................................... 161
Acknowledgements . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 161
References . . . . ...................................................................................... 161

1. Introduction

Autism-spectrum disorders (ASD) are lifelong neurodevelopmental disorders characterised by impairments in communication, reciprocal
social interaction, and restricted and repetitive behaviours and interests (WHO, 1992). ASD affects at least 1% of the population (Brugha et al.,
2011), although there is substantial heterogeneity in the ASD symptom profile and hence the level of resultant impairment. Individuals with ASD
commonly experience co-morbid mental health conditions including anxiety and affective disorders (Joshi et al., 2013; NICE, 2012; Simonoff et
al., 2008), and ‘‘emotional and behavioural problems’’ including anger, disrupted sleep, and restricted eating (Maskey, Warnell, Parr, Le Couteur,
& McConachie, 2013). Psychiatric co-morbidity can compound difficulties with social and occupational functioning, as well as attainment of
independent living skills. Co-morbidity is also associated with a significantly increased carer burden ( Cadman et al., 2012; Karst & Van Hecke,
2012), highlighting the need for the development of targeted treatments (NICE, 2012).

1.1. Cognitive behaviour therapy

For the typically-developing population, cognitive behaviour therapy (CBT) interventions are used routinely to treat a wide range of mental
health disorders (NICE, 2011), as well as associated transdiagnostic characteristics such as insomnia (Vitiello, McCurry, & Rybarczyk, 2013) and
worry (Covin, Ouimet, Seeds, & Dozois, 2008). CBT is a type of talking therapy (psychotherapy), which primarily aims to help individuals to
(1) notice and understand how their thoughts, behaviours and emotions are inter-related and (2) develop new ways of thinking about, coping with
and responding to, anxiety-provoking or distressing situations (Beck, 2011). CBT is a short-term goal-orientated approach: individuals are
encouraged to identify specific measurable goals they would like to work towards in order to attain symptom reduction. In line with Stepped
Care approaches to mental health service provision, for example the UK Improving Access to Psychological Therapies Initiative (IAPT) (Clark,
2011; NICE, 2011) CBT is increasingly being delivered through novel modalities including guided self-help, computerised packages, and group-
based formats. The therapist’s tool-box is also expanding: interventions now commonly utilised range from traditional behavioural and cognitive
techniques, to ‘third wave ’
approaches, for example metacognitive, mindfulness-based, and acceptance and
commitment (ACT) techniques.
 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162 153

1.2. Cognitive behaviour therapy for individuals with ASD

It is hypothesised that the structure, process and content of standard CBT require adaptation for the ASD population for several reasons
(Anderson & Morris, 2006; Attwood, 2004; Gaus, 2011). First, core ASD characteristics such as socio-communication deficits and difficulties
tolerating change and uncertainty can affect engagement with the therapist and the therapy process. Second, alexithymia, i.e. difficulties with
describing and labelling emotions, commonly co-exists with ASD (Bird & Cook, 2013); hence individuals may find it hard to identify links
between cognitions (thoughts and beliefs), emotions and resultant behavioural responses, all of which are integral to the development of a shared
formulation of the presenting problem. Third, neuropsychological impairments commonly associated with ASD, such as deficits in theory of
mind and cognitive flexibility, and weak central coherence (a preference for local versus global processing) ( Brunsdon & Happe, 2014) may
affect an individual’s ability to utilise standard cognitive and behavioural techniques. For example, they may experience difficulties with
generating alternative or additional perspectives for challenging negative thoughts, or be unable to generalise the findings from behavioural
experiments and exposure-based tasks undertaken during sessions. Consequently, several adaptations are proposed to be pertinent for enhancing
engagement, acceptability and utility of CBT approaches. These include (1) use of written and pictorial methods to enhance discussion during
assessment, therapy and to facilitate recall; (2) identification of idiosyncratic descriptions of emotions (e.g. anxiety or low mood); (3) tailor-made
individualised outcome measures (e.g. analogue scales); (4) enhancement of emotional literacy prior to ‘active’ CBT treatment; (5) emphasis on
behavioural change and skills development; and (6) a less socratic therapeutic style (see also Anderson & Morris, 2006; Attwood, 2004; Gaus,
2011).

1.3. Rationale for this review

Several randomised controlled trials (RCTs) have investigated the effectiveness of modified CBT for children and adolescents with ASD and
co-morbid anxiety disorders (e.g. Reaven, Blakeley-Smith, Culhane-Shelburne, & Hepburn, 2012; Sofronoff, Attwood, & Hinton, 2005;
Sofronoff, Attwood, Hinton, & Levin, 2007; White et al., 2013; Wood, Drahota, Sze, Har, Chiu, & Langer, 2009). Overall, reviews of the
empirical data indicate that CBT can be effective for reducing anxiety symptoms and improving functioning in young people with ASD (Danial
& Wood, 2013; Lang, Regester, Lauderdale, Ashbaugh, & Haring, 2010). Generalisability of study findings to other young and adult ASD
populations, however, is hampered by methodological limitations such as heterogeneity in participants’ clinical presentations, small sample sizes,
variability in outcome measures used, and lack of independent assessment. With regard to the effectiveness of CBT for adults with ASD, a recent
literature review – which focused on CBT for core and co-morbid symptoms, and included studies published until 2010 – highlighted the dearth
of high quality studies in this area (Binnie & Blainey, 2013).

Thus far, it has not been established whether certain types of CBT interventions – for example behavioural approaches versus cognitive
techniques – are more effective in treating co-morbid mental health disorders and characteristics in adults with ASD. Similarly, the extent to
which CBT interventions for co-morbidities are delivered via standard disorder-specific protocols (NICE, 2011) or deviate from these, is not
certain, yet this has important implications for clinical service provision.
Hence as an update to existing reviews and using a broader frame of reference, the aims of this review were (1) to determine whether CBT
interventions are an effective treatment for psychiatric disorders and transdiagnostic mental health symptoms in adults with ASD; (2) to
determine whether certain treatment modalities or interventions have been more commonly used to target co-morbid symptoms; and if so,
whether any one of these were considered more effective, and (3) to outline clinical practice implications arising from the literature.

2. Methods

2.1. Search strategy

A search strategy was registered with the International Prospective Register of Systematic Reviews (Prospero: ID CRD42013005665) ( Spain,
Sin, Chalder, Murphy, & Happe, 2013). We searched the Medline, PsychInfo and Embase databases for studies published between 1st January
1993 and 31st August 2013: this time frame was chosen as CBT has become more routinely introduced across health care clinical settings since
the mid-1990s. A supplementary database search was also undertaken in Web of Science, PubMed, and the Cochrane Central Register of
Controlled Trials (CENTRAL). A combination of search terms was used including: autis* – autis* spectrum disorder* – Asperger* –
development* disorder* and cognitive behavio* therap* – behavio* therap* – cognitive therap* – talking therap* – third wave approaches –
psychological intervention* – treatment* – mindfulness – ACT – metacognitive therap* – self help. No stipulation was made about comparator
interventions or types of outcomes, in order to maximise the scope of the search.

2.2. Inclusion criteria

Pre-determined study inclusion criteria were (1) papers published in peer-reviewed English-language journals; (2) primary research including
case studies, case series, quasi-experimental studies or RCTs; (3) designs that sought to test the effectiveness of CBT interventions for
psychiatric co-morbidity or associated mental health characteristics; (4) individuals
154 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162

aged 18 and over, diagnosed with an ASD (including autism, Asperger’s syndrome, atypical autism and pervasive developmental disorders); (5)
studies using any modality of treatment e.g. individual sessions or group-based therapy; and
(6) studies employing at least one pre- and post-treatment self-, informant-, or clinician-rated outcome measure relevant to mental health or
functioning.

2.3. Exclusion criteria

Non-English language publications and grey literature were excluded. We also excluded studies that described interventions primarily offered
to target core ASD symptoms, such as social skills training or enhancement of social cognition. Studies which focused solely on CBT
interventions delivered to children and adolescents with ASD were omitted, although we allowed for the possibility of contacting authors for
sub-group data i.e. studies that combined young people and adults.

2.4. Study selection and data extraction

The database searches and study selection were undertaken by two people (see Fig. 1). A total of 1190 articles were retrieved initially; 364
duplicates were removed at this stage. The titles and abstracts of 826 articles were reviewed by one author (DS). To enhance rigour, 10% (84
papers) of all titles discounted at this stage and chosen at random, were independently reviewed by a second author (JS), and agreement was
reached in all cases. Twenty-four articles were retrieved for full text review and screened independently by two authors. Of these, 18 articles did
not meet the review criteria (reasons are outlined in Fig. 1). Based on joint consensus, they were excluded at this stage.

A total of six articles met the review criteria. Reference lists of these six papers, as well as the existing literature review ( Binnie & Blainey,
2013) were hand searched; no new studies were found for inclusion. A data extraction form, adapted from NICE guidance ( NICE, 2006), was
used to summarise data about the following factors: (1) study methods and setting; (2)

Potentially relevant papers identified in search

(n = 1190)

Duplicates removed (n = 364)

Papers excluded based on reading title and abstracts
(n = 802)

Papers retrieved for full text review (n = 24)

+
Reference lists of pre-existing reviews (n = 0)
+
Hand search of reference lists of studies included (n = 0)

Papers excluded
(n= 18 studies, 18 papers)
excluded due to population (n = 12 ), intervention (n = 4),
outcome (n = 1 ), trial in process, no outcome data available
(n = 1)

Quantitative studies included

(n = 6 studies; 6 papers)
RCTs (n = 2); quasi-experimental designed studies (n = 1);
case series (n = 1); case studies (n = 1)

Fig. 1. Systematic search results.

 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162 155

population demographics, clinical and diagnostic details; (3) type, modality, frequency and duration of treatment; (4) outcome measures; (5)
treatment outcomes; and (6) factors associated with treatment fidelity.

2.5. Plan for analysis

Due to the significant heterogeneity between participant characteristics and outcome measures used, it was not possible to pool the data and
undertake a meta-analysis. Therefore, a narrative method of analysis was undertaken.

3. Results

3.1. Study methods and setting

Table 1 summarises information about studies included in the review. Six papers met the review inclusion criteria: two RCTs (Russell et al.,
2013; Spek, van Ham, & Nyklicek, 2013) one quasi-experimental study (Russell, Mataix-Cols, Anson, & Murphy, 2009), one group-based case
series intervention (Weiss & Lunsky, 2010), and two case studies (Cardaciotto & Herbert, 2004; Hare, 1997). One study (Russell et al., 2013)
included both adults and adolescents with ASD; the first author was contacted regarding sub-group data for the adult sample, and hence this
study is included in our review. One study was undertaken in Sweden ( Spek et al., 2013), three in the UK (Hare, 1997; Russell et al., 2009,
2013), and two in North America (Cardaciotto & Herbert, 2004; Weiss & Lunsky, 2010). A combined total of 105 adults with ASD participated
in the studies.

3.2. Quality assessment

No studies were excluded based on quality assessment. It was acknowledged that there were potential biases – such as sampling, selection
and measurement bias (Coolican, 2009) – inherent to the case studies (Cardaciotto & Herbert, 2004; Hare, 1997), the case series (Weiss &
Lunsky, 2010), and the quasi-experimental study (Russell et al., 2009). The RCTs by Russell et al. (2013) and Spek et al. (2013) employed
several methods to enhance internal study validity including independent random sequence allocation of participants to test or control conditions,
blinding of assessors who supported participants to complete outcome measures, and use of intention to treat (ITT) data analysis methods.
Therapists and participants in both studies were not blinded to treatment, given that the comparator interventions were an active treatment
(anxiety management; Russell et al., 2013) and a wait list control (Spek et al., 2013).

3.3. Participant demographics

Both case studies (Cardaciotto & Herbert, 2004) involved male participants. The group-based case series, quasi-experimental study and two
RCTs (Russell et al., 2009, 2013; Spek et al., 2013; Weiss & Lunsky, 2010) included both males and females. Across studies, there was a higher
proportion of male participants and it is unclear whether this was due to sampling and selection strategies, the over-representation of ASD in
males (Baron-Cohen et al., 2011), or other factors. The ages of participants across studies ranged from 18 to 65 years. Mean ages of participants
were dissimilar for the RCTs; individuals in the Spek et al. (2013) study were considerably older than those who took part in the study by Russell
et al. (2013). Overall, there were limited data about participants’ ethnicity or socio-economic backgrounds.

3.3.1. Participant clinical diagnoses

All participants were reported to have diagnoses of ASD, including high-functioning autism, Asperger syndrome or pervasive developmental
disorder (PDD). In three studies, ASD diagnosis was confirmed via use of standardised assessment methods such as ICD-10 clinical diagnostic
criteria (WHO, 1992), the Autism Diagnostic Interview – revised (ADI-r) (Lord, Rutter, & Lecouteur, 1994) or the Autism Diagnostic
Observation Schedule – generic (ADOS-g) (Lord et al., 2000; Russell et al., 2009, 2013; Spek et al., 2013), although not all participants
underwent all assessments. ASD diagnosis was not confirmed independently for one case study (Hare, 1997) and the group-based intervention
(Weiss & Lunsky, 2010). For the second case study (Cardaciotto & Herbert, 2004), diagnosis was confirmed using the Asperger Syndrome
Diagnostic Interview (ASDI) (Gillberg, Gillberg, Rastam, & Wentz, 2001). The age at which ASD was diagnosed was not specified for most
participants.

3.3.2. Co-morbid psychiatric diagnosis

Participants were reported to have a range of co-morbid mental health conditions, including social anxiety disorder (Cardaciotto & Herbert,
2004), obsessive-compulsive disorder (OCD) (Russell et al., 2009, 2013), symptoms associated with low mood or depression (Hare, 1997;
Russell et al., 2013; Spek et al., 2013; Weiss & Lunsky, 2010), self-harm (Hare, 1997), post-traumatic stress disorder (PTSD) (Weiss & Lunsky,
2010), agoraphobia (Weiss & Lunsky, 2010), and non-specific anxiety (Russell et al., 2013; Spek et al., 2013; Weiss & Lunsky, 2010). Diagnosis
of co-morbidity was established using ICD-10 (WHO, 1992) or DSM-IV criteria (First, Spitzer, Gibbon, & Williams, 2002) (Cardaciotto &
Herbert, 2004; Russell et al., 2009, 2013), clinical impression at assessment (Hare, 1997; Spek et al., 2013; Weiss & Lunsky, 2010), and
independent formal assessment (Russell et al., 2013).
Limited data were available about the age of initial onset of mental
health co-morbidity, or the duration and trajectory of these symptoms.
156 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162
Table 1

Overview of studies.

Study Method Sample (n, core and Intervention Outcome measures Results
co-morbid diagnoses)
Cardaciotto and Case study 1 male Number of sessions: 14 SPAI Improvement in social
Herbert (2004) anxiety and low mood
US symptoms
26 years old LSAS
Asperger syndrome Session duration: not BDI-II
specified CGI
Social phobia, Techniques: social Behavioural
depressive symptoms skills training, skills assessment
rehearsal (role plays),
cognitive restructuring
Hare (1997) Case study 1 male Number of sessions: 17 BDI Improvement in low

UK mood symptoms
23 years old
Asperger syndrome Session duration: 30– Idiosyncratic Reduced frequency of
60 min record of self-harm self-harm
incidents
Low mood, self-harm, Techniques: emotional
excessive alcohol use literacy, graded
exposure, cognitive
restructuring,
relaxation and
distraction
Russell et al. (2009) Quasi- 21 males, 3 females Number of sessions: 10– YBOCS 58% (n = 7) improved

UK experimental, Mean age 23.8 (CBT) 50 session (mean 27.5) with CBT compared
non- Mean age 32.1 (TAU) with 16% (n = 2) TAU
randomised group
trial, CBT for
OCD (n = 12) vs
TAU (n = 12)
ASD diagnosis Session duration: not BDI
specified
OCD Techniques: exposure- BAI OCD symptom
based tasks, cognitive reduction more likely
restructuring for CBT group
Symptoms of anxiety No statistically
or low mood significant changes or
differences in BDI and
BAI scores, between
groups
Russell et al. RCT 35 adults Number of sessions: YBOCS Improvements in OCD

(2013) UK Mean age 28.6 (CBT) Mean CBT: 17.4 symptoms reported for
BDI CBT and AM
conditions, 45%
CBT for OCD Mean age 25.2 (AM) Mean AM: 14.4 BAI treatment responders
(n = 20) vs CGI (CBT) and 20%
anxiety treatment responders
management (AM)
(AM) (n = 17) ASD diagnosis Session duration: 1 h WSAS
Manualised OCD diagnosis Techniques: emotional No significant
approach literacy, exposure- differences between
Symptoms of anxiety, based tasks, cognitive groups pre- and post-
or low mood restructuring treatment on other
measures
Spek et al. (2013) RCT 27 males, 14 females Number of sessions: 9 SCL-90-R Improvements in

Sweden Mean age 44.4 (MBSR) RRQ mood, rumination and

Mindfulness- Mean age: 40.1 (WL) Session duration: 2.5 h DGMS, positive anxiety symptoms
based group affect sub-scale following MBSR
(MBSR) (n = 20) compared to wait list
vs wait list (WL) control, increase in
(21) positive affect
ASD diagnosis Techniques: Differences between
Mindfulness-based groups, Cohen’s d 0.76
techniques including (anxiety) and 1.25
meditation (rumination)
Manualised Symptoms of anxiety,
approach depression or
rumination
 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162 157
Table 1 (Continued )

Study Method Sample (n, core and Intervention Outcome measures Results
co-morbid diagnoses)
Weiss and Group-based 2 males, 1 female Number of sessions: 12 BDI-II Downward trend in

Lunsky (2010) case series anxiety symptoms, no

US significant change in
self-report of mood
pre- and post-
treatment
Manualised Asperger syndrome Session duration: BAI
approach 60 min
Depression, PTSD, Techniques: emotional
agoraphobia literacy, cognitive
restructuring,
behavioural
experiments,
behavioural
experiments

TAU – treatment as usual, BDI – Beck Depression Inventory, BAI – Beck Anxiety Inventory, SPAI – Social Phobia and Anxiety Inventory, LSAS – Liebowitz Social Anxiety
Scale, CGI – Clinical Global Impression, YBOCS – Yale-Brown Obsessive Compulsive Scale, SCL-90-R – Symptom checklist revised, RRQ – Rumination Reflection
Questionnaire, DGMS – Global Mood Scale (Dutch version), WSAS – Work and Social Adjustment Scale.

3.4. Structure and content of CBT treatment

3.4.1. CBT modality

Participants were offered CBT via individual sessions (Cardaciotto & Herbert, 2004; Russell et al., 2009, 2013) or group-based approaches
(Spek et al., 2013; Weiss & Lunsky, 2010). None of the studies reported using a combined individual and group-based approach.

3.4.2. Manualised treatment protocols

The case studies and quasi-experimental study did not use a formulaic approach. Conversely, the group-based case series (Weiss & Lunsky,
2010) and the RCTs (Russell et al., 2013; Spek et al., 2013) were informed by a treatment manual. Spek et al. (2013) for example, utilised a
mindfulness-based protocol adapted to exclude some of the cognitive elements of treatment, and to increase the duration of some experiential
tasks. Russell et al. (2013) described basing their manual on prior pilot work, clinical records review and the literature, as well as more recent
standard CBT protocols used for treating OCD in the typically-developing population.

3.4.3. CBT techniques

Studies employed a range of CBT techniques. Excluding the RCT by Spek et al. (2013) which was principally a mindfulness intervention,
each of the studies incorporated both cognitive and behavioural techniques. Interventions described included
(1) exposure-based work whereby participants identified and worked through a hierarchy of difficult situations (Hare, 1997; Russell et al., 2009,
2013); (2) relaxation techniques (Hare, 1997); (3) distraction techniques (Hare, 1997; 4) social skills training (Cardaciotto & Herbert, 2004); (5)
skills rehearsal (role plays) (Cardaciotto & Herbert, 2004); and (5) identification, examination and restructuring of negative thoughts and beliefs
(Cardaciotto & Herbert, 2004; Russell et al., 2009; Weiss & Lunsky, 2010). Several studies also reported including a psycho-educational
component about emotions or anxiety (Weiss & Lunsky, 2010), or rumination (Spek et al., 2013). The RCT by Spek et al. (2013) comprised
mindfulness-based techniques; specifically, enhancing awareness and learning increasingly sophisticated meditation practices. The active
comparator in the Russell et al. (2013) study was anxiety management, and mainly consisted of psycho-education, relaxation and distraction
techniques, and skills-based approaches (problem-solving).

3.4.4. Frequency and duration

The number of sessions attended by participants across the studies ranged from 9 (Spek et al., 2013) to 50 (although the mean number of
sessions was 27.5 for this study, Russell et al., 2009). Sessions were primarily offered on a weekly basis. Several studies described either
increasing the number of assessment sessions to aid with engagement or socialisation to the CBT model (e.g. Hare, 1997; Russell et al., 2013) or
to accommodate the impact of core ASD characteristics or associated neuropsychological impairments, such as difficulties with information
processing (e.g. Russell et al., 2009, 2013; Spek et al., 2013). Session duration ranged from 30 min (Hare, 1997) to 2.5 h (Spek et al., 2013);
three studies reported that sessions were around 1 h on average (Russell et al., 2009; Weiss & Lunsky, 2010).

3.4.5. Homework
Each of the studies described implementing homework as standard. This mainly included practising behavioural and cognitive strategies
acquired during sessions, as well as provision of bibliotherapy materials (e.g. Weiss & Lunsky, 2010). The
158 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162

amount and type of homework tasks for the two group-based interventions (Spek et al., 2013; Weiss & Lunsky, 2010) appeared to be fairly
prescribed, i.e. this corresponded to the session outlines reported. Limited data were available about participant compliance with suggested
homework, although Russell et al. (2013) stated that trial therapists utilised a more proactive approach in planning homework tasks and that,
overall, study participants completed significantly more behavioural than cognitive tasks.

3.4.6. Adaptations to CBT

Studies described introducing several modifications to the process and content of standard CBT. First, the average number of sessions offered
to treat specific disorders, notably OCD (Russell et al., 2009, 2013) and low mood (Hare, 1997) exceeded UK NHS treatment guidelines (NICE,
2011). Additionally, the mindfulness-based group RCT (Spek et al., 2013) increased the session number slightly from the standard protocol.
Second, several studies (Hare, 1997; Russell et al., 2009, 2013) emphasised the deliberate inclusion of materials, from the outset of treatment, to
enhance participant understanding of emotions and anxiety. Third, several authors described modifying the means through which information
was delivered, such as using additional visual cues and written materials, avoidance of colloquialisms, and concerted attempts to identify specific
and concrete examples. Fourth, there was some accommodation of core ASD characteristics during and outside of CBT sessions. Spek et al.
(2013), for example, appeared to slow down the rate at which information was discussed, and also increased the duration of some experiential
tasks, to overcome potential cognitive processing deficits. Russell et al. (2009, 2013) sought to incorporate participants’ interests to enhance
engagement and facilitate learning of new techniques. Also, Russell et al. (2013) and Spek et al. (2013) reported efforts to increase compliance
with homework, adopting a more ‘structured approach’. Finally, several studies described that therapists were more directive, i.e. less socratic in
their style than would typically be the case in CBT.

3.5. Outcomes

3.5.1. Outcome measurement

All studies utilised self-report questionnaires to assess change and treatment outcomes completed pre- and post-treatment or more regularly.
Questionnaires measured a range of mental health symptoms: low mood (Beck Depression Inventory: BDI) ( Beck, Ward, Mendelson, Mock, &
Erbaugh, 1961), positive affect (Dutch Global Mood Scale, positive affect sub-scale: DGM) (Denollet, 1993), social anxiety ((Liebowitz Social
Anxiety Scale: LSAS) (Liebowitz, 1987) and Social Phobia Anxiety Inventory: SPAI) (Turner, Beidel, Dancu, & Stanley, 1989), anxiety (Beck
Anxiety Inventory: BAI) (Beck, Epstein, Brown, & Steer, 1988), OCD (Obsessive compulsive inventory – revised: OCI-r) (Foa et al., 2002),
rumination (Rumination-Reflection Questionnaire: RRQ) (Trapnell & Campbell, 1999), and general distress (Dutch version of the Symptom
Checklist – 90 – revised: SCL-90-r) (Derogatis, 1994). One study (Russell et al., 2013) measured participants’ functioning with the Work and
Social Adjustment Scale (Mundt, Marks, Shear, & Greist, 2002), as well as satisfaction with treatment. Two studies (Cardaciotto & Herbert,
2004) sought objective opinions of change using the Clinical Global Impression scale (CGI) ( Guy, 1976). Additionally, two studies (Russell et
al., 2009, 2013) included a clinician-administered measure of OCD symptoms (the Yale-Brown Obsessive Compulsive Scale: YBOCS)
(Goodman et al., 1989) and one study included independent observation of participant behaviour (Cardaciotto & Herbert, 2004).

The most commonly used questionnaires were the BDI (Beck et al., 1961; Cardaciotto & Herbert, 2004; Russell et al., 2009, 2013, Weiss &
Lunsky, 2010) and the BAI (Beck et al., 1988; Russell et al., 2009; Weiss & Lunsky, 2010). It was noted that while questionnaires administered
have good psychometric properties for the typically-developing population, none have been validated for use with individuals with ASD.
Overall, there were limited data available about whether the suggested normative thresholds for clinical caseness appeared to apply to study
participants, or tallied across multiple measures.

3.5.2. Treatment outcomes – effectiveness of CBT

A decrease in co-morbid mental health symptoms was reported for all studies. For the case studies, there were reported improvements in
mood (Cardaciotto & Herbert, 2004), and some improvements in social skills and concerns (Cardaciotto & Herbert, 2004). Weiss and Lunsky
(2010) noted that the three participants involved in their group-based treatment benefited from psycho-education and information about CBT;
self-report scores of mood and anxiety symptoms fluctuated during the intervention, with similar ratings of mood pre- and post-treatment, and a
downward trend in self-ratings of anxiety. For the quasi-experimental study, Russell et al. (2009) described that individuals offered CBT versus
treatment as usual (for OCD) were (1) more likely to experience symptom reduction, and (2) that 58% of participants (n = 7) offered CBT
attained reduction in both obsessions and compulsions. There were also some noted reductions in self-report scores of anxiety symptoms and low
mood, although the differences between the groups were not statistically significant. In the RCT comparing CBT versus anxiety management for
OCD, Russell et al. (2013) found that both interventions were of benefit for a proportion of participants: overall CBT was considered to be
also found that participants who were offered the active
associated with higher rates of treatment response. Spek et al. (2013)
intervention (a mindfulness group) showed improvements in mood and anxiety symptoms, compared to the
comparator (wait list) group.
 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162 159

3.6. Attrition

Attrition rates were considered for the two RCTs (Russell et al., 2013; Spek et al., 2013). One participant (2.4%) dropped out of active
intervention group in the study by Spek et al. (2013); the reason for this was reported as illness. Russell et al. (2013) reported that there were six
drop outs (13%), three from the CBT group and three in the control group (anxiety management).

3.7. Fidelity to treatment

Therapists involved in the RCTs (Russell et al., 2013; Spek et al., 2013), the quasi-experimental study (Russell et al., 2009) and the group-
based intervention (Weiss & Lunsky, 2010) were reported to have undertaken specialist training around working with individuals with ASD and
clinical co-morbidity. Of these studies, all bar the quasi-experimental study followed a manualised approach. Fidelity was also maintained in one
study (Russell et al., 2013) through independent ratings of randomly selected audio-recordings of sessions.

4. Discussion

4.1. Overview

This systematic review aimed to summarise published empirical data regarding the effectiveness of CBT interventions for co-morbid mental
health conditions in adults with ASD. Six of 18 potentially relevant studies met the pre-determined review inclusion criteria. The quality of four
studies (two case studies, one group-based case series, and a quasi-experimental study) could not be appraised comprehensively, due to the
designs and methods. Notwithstanding this, several participants in these studies were reported to have benefited, clinically, from CBT
interventions, as noted by a reduction in symptom severity scores on self-report questionnaires and, in one case, on a clinician-administered
measure of OCD (Russell et al., 2009). Only two studies were undertaken using more rigorous RCT conditions; one study sought to treat co-
morbid OCD symptoms using both behavioural and cognitive interventions (Russell et al., 2013), and one RCT sought to reduce low mood and
rumination, and enhance positive affect via a group-based mindfulness approach (Spek et al., 2013). A proportion of participants involved in the
RCTs attained global improvements, in terms of a decrease in co-morbid mental health symptoms, improvement in functioning, or an increase in
positive affect.

The review findings are broadly consistent with an earlier review (of studies up to 2010) which focused on CBT for the adult ASD
population, but not psychiatric co-morbidity specifically (Binnie & Blainey, 2013). We were able to include three additional studies of CBT for
co-morbid mental health conditions (Russell et al., 2013; Spek et al., 2013; Weiss & Lunsky, 2010), two of which were RCTs and therefore
perhaps provide more robust evidence. Our findings were also consistent with previous reviews of CBT for children and adolescents with ASD
(e.g. Danial & Wood, 2013; Lang et al., 2010). Lang, Regester, Lauderdale, Ashbaugh, and Haring (2010), for example, reviewed the
effectiveness of CBT for anxiety delivered to a total of 110 participants (only one of whom was reported to be an adult), across nine studies.
They found that CBT techniques had utility for the ASD population, albeit that there was wide variation in the number of sessions offered (6–16
sessions), the duration of therapy (6–26 weeks) and the weighting of behavioural versus skills-based versus cognitive techniques employed
(Lang et al., 2010). With the broader remit of this review to include the wide variation of techniques that fall under the umbrella term CBT (and
are typically delivered as part of CBT service delivery), we found preliminary evidence that mindfulness-based approaches may also be of
benefit for individuals with ASD.

Despite the reported high rates of anxiety, affective and behavioural disorders in the adult ASD population (Hofvander et al., 2009; NICE,
2012), relatively few studies have thus far been undertaken to evaluate the effectiveness of non-pharmacological interventions to treat co-morbid
psychiatric symptoms. This is of concern given that (in primary health care settings) psychotropic drugs are prescribed to approximately 30% of
ASD individuals (and of these many have more than one prescription) (Murray et al., 2014). The clinical implication is that adults with ASD may
either fail to be offered formal help, or may be offered sub-optimal treatments that neither have demonstrated efficacy, nor take into account the
potential impact of core ASD characteristics or associated impairments on engagement with therapists and the therapy process. This appears to
be in stark contrast to recent initiatives which have sought to increase the availability and accessibility of psychological interventions – in
particular CBT – for the typically-developing population (Clark, 2011; Gyani, Shafran, Layard, & Clark, 2013), including individuals with long-
term conditions (e.g. Wroe, Rennie, Gibbons, Hassy, & Chapman, 2014).

4.2. Limitations

Several limitations to the review should be noted. First, while we undertook the search in several databases, we excluded non-English
language publications due to resource constraints. Second, although we did not exclude co-morbid intellectual disability, we did not search for
papers that were primarily about CBT undertaken with this clinical population, some of whom may also have diagnoses of ASD. Third, while we
searched the published literature systematically, we were not able to account for publication bias, e.g. retrospectively searching trials registers for
any potentially unpublished studies, or those which may have had equivocal findings.
160 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162

4.3. Clinical practice implications

While only six studies are included within this review, it is nonetheless pertinent to consider the implications for clinical practice and service
provision. First, it may be beneficial for CBT clinicians to be aware of the severity of an individual’s core ASD characteristics across the three
domains (reciprocal social interaction, communication, and restricted and repetitive behaviours and interests), as well as potential co-occurring
alexithymia or impairments in neuropsychological functioning, for example theory of mind ability and information processing styles, and
resistance to change. Such information is likely to inform decision-making about how best to undertake the assessment, what interventions to
use, and in which order (e.g. behavioural versus cognitive versus skills-based techniques). Objective knowledge about the extent and range of
these impairments may encourage clinicians to adopt a modified therapeutic approach, e.g. a more didactic style, in order to maximise
engagement. Similarly, objective assessment of alexithymia and inclusion of emotional literacy sessions may be useful at the outset of a course
of CBT, in order to provide individuals with ASD with the requisite knowledge and language to be able to engage in subsequent conversations
about physiological arousal and emotions.

Second, use of objective as well as idiosyncratic (individualised) ratings of mental health symptoms pre-, mid- and post-treatment seems an
essential approach to diagnosing associated symptoms in clinical settings. Participants across studies included in our review were diagnosed with
a range of co-morbid mental health conditions, most frequently, OCD, anxiety disorders and low mood. Clinical diagnoses for study participants
were established using standardised diagnostic criteria, clinician-rated assessment, and/or clinical impression. However, diagnoses of psychiatric
co-morbidity in ASD is notoriously complex; particularly in the case of OCD (Russell, Mataix-Cols, Anson, & Murphy, 2005) and social anxiety
(Tyson & Cruess, 2012) whereby there are significant overlaps in the core and co-morbid symptom profiles. Also, in our data analysis, it proved
difficult to determine conclusively the proportion of participants who may have experienced symptoms of several clinical disorders concurrently,
or presented with clinically meaningful but sub-threshold symptoms. Given the heterogeneous nature of ASD, diagnostic overshadowing
between disorders, and potential difficulties with introspection that individuals with ASD can experience, reliance on one method of assessment
may incur either false-positive or false-negative results. Also, given that individuals with ASD can experience heightened anxiety in novel
situations, it may be that several baseline assessments are prudent, for example, to reduce the likelihood of individuals acquiescing in response to
clinician questions or self-report questionnaires; or in order to adequately assess the full range of symptoms that individuals may have.

Third, it is important that clinicians are mindful about the types of self-report questionnaires they utilise to measure treatment outcomes and
change. Self-report questionnaires of mood and anxiety typically include items about physiological arousal and emotions, and sometimes have
reversed-scored items. Whether these items are meaningful for individuals with ASD, e.g. due to alexithymia, should not be presupposed. Also,
while each of the studies in our review used widely administered questionnaires with good psychometric properties, their reliability and validity
for the ASD population is yet to be established (e.g. Lecavalier et al., 2013).

Fourth, there are several integral features of CBT that may be well suited for the ASD population. CBT is a highly structured approach. There
are clear goals for treatment and commonly used interventions such as behavioural exposure and behavioural experiments are planned and
undertaken in a graded manner. The implication is that therapy is somewhat predictable, and limits the element of surprise or uncertainty that
people with ASD can find hard to tolerate. Also a key strength of CBT is that clinicians have a metaphorical tool-box, comprised of different
techniques that can be applied to specific disorders and transdiagnostic symptoms (e.g. sleep or eating difficulties) at a pitch and pace that suits
each patient. While protocol-driven manualised CBT is the favoured approach when working with less complex individuals (e.g. NICE, 2011),
the clinical implication is that a more flexible style is likely needed when working with individuals who have multiple mental health symptoms,
and long-term, entrenched difficulties, as is often the case for adults with ASD. This may include a longer assessment phase and an increased
number of treatment sessions to aid with initial engagement with the therapist, to enhance emotional literacy, and to practice, consolidate and
generalise techniques learnt. While this may incur additional financial and resource implications, it could be hypothesised that these costs are
offset by improved outcomes.

4.4. Implications for research

There are several implications for research. First, there is a small but emerging body of evidence to suggest that psychological interventions,
in particular CBT, may have utility in treating co-morbid mental health symptoms in adults with ASD. But there is now a pressing need for
larger, more rigorous trials that investigate the effectiveness and acceptability of CBT for (1) specific mental health conditions (e.g. anxiety
disorders and depression); (2) transdiagnostic characteristics commonly associated with psychiatric co-morbidity (e.g. anger and sleep
difficulties); and (3) using different mediums of delivery (e.g. individual sessions versus group interventions versus self-help approaches).
Second, clinical consensus is that the content and structure of CBT require modification to enhance outcomes (e.g. Attwood, 2004; Anderson and
Morris, 2006; Gaus, 2011). Yet research is needed to better understand mediating and moderating factors for CBT in ASD i.e. what are the
essential ingredients of the therapy process and content, and how can interventions be modified to best
accommodate inherent ASD traits and associated neuropsychological impairments. Finally, it is important that
there is increased research into establishing reliable and valid outcome measures, either through (1) determining
normative thresholds (for the ASD population) for commonly used psychopathology measures, and/or (2)
developing questionnaires and psychophysiological
 D. Spain et al. / Research in Autism Spectrum Disorders 9 (2015) 151–162 161

measures tailored to the specific needs of individuals with ASD, for example those that include response items that are unambiguous and rely
less on self-appraisal of affect and emotions.

5. Conclusion

Individuals with ASD are commonly considered to be at risk of developing co-morbid mental health conditions. To date, however, studies
investigating causes and consequences of, and treatments for psychiatric co-morbidity, have primarily focused on children and adolescents with
ASD. This review has sought to synthesise the data from published empirical studies about CBT for adults with ASD; the most widely evidence-
based treatment for anxiety and affective disorders in typically-developing adult populations. The review findings indicate that CBT shows
promise, but that adaptations are likely needed to augment the effectiveness and acceptability of standard interventions. Formally developing the
evidence-base about the mediating and moderating mechanisms (for CBT for adults with ASD) is an important next step for clinicians and
researchers.

Acknowledgments

DS is funded by a National Institute for Health Research (NIHR) Clinical Doctoral Research Fellowship (CDRF-2012-03-059). JS is funded
by a NIHR Doctoral Research Fellowship (DRF-2011-04-129). The review presents independent research funded by the NIHR. The views
expresed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health. TC is part supported by the
NIHR Biomedical Research Centre for Mental Health at the South London and Maudsley NHS Foundation Trust and Institute of Psychiatry,
King’s College London. We thank the National Institute for Health Research Biomedical Research Centre for Mental Health at King’s College
London, Institute of Psychiatry, and South London and Maudsley National Health Service Foundation Trust, the NIHR Crossing the Divide
Programme Study, and the Dr Mortimer and Theresa Sackler Foundation for their financial support. Acknowlegements to Mr John Gale,
Librarian, for advice about the search strategy.

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