Mediation of 6-year mid-childhood follow-up outcomes after pre-school social communication (PACT) therapy for autistic children: randomised controlled trial.

BACKGROUND: There are very few mechanistic studies of the long-term impact of psychosocial interventions in childhood. The parent-mediated Paediatric Autism Communication Therapy (PACT) RCT showed sustained effects on autistic child outcomes from pre-school to mid-childhood. We investigated the mechanism by which the PACT intervention achieved these effects. METHODS: Of 152 children randomised to receive PACT or treatment as usual between 2 and 5 years of age, 121 (79.6%) were followed 5-6 years after the endpoint at a mean age of 10.5 years. Assessors, blind to the intervention group, measured Autism Diagnostic Observation Scale Calibrated Severity Score (ADOS CSS) for child autistic behaviours and Teacher Vineland (TVABS) for adaptive behaviour in school. Hypothesised mediators were child communication initiations with caregivers in a standard play observation (Dyadic Communication Measure for Autism, DCMA). Hypothesised moderators of mediation were baseline child non-verbal age equivalent scores (AE), communication and symbolic development (CSBS) and 'insistence on sameness' (IS). Structural equation modelling was used in a repeated measures mediation design. RESULTS: Good model fits were obtained. The treatment effect on child dyadic initiation with the caregiver was sustained through the follow-up period. Increased child initiation at treatment midpoint mediated the majority (73%) of the treatment effect on follow-up ADOS CSS. A combination of partial mediation from midpoint child initiations and the direct effect of treatment also contributed to a near-significant total effect on follow-up TVABS. No moderation of this mediation was found for AE, CSBS or IS. CONCLUSIONS: Early sustained increase in an autistic child's communication initiation with their caregiver is largely responsible for the long-term effects from PACT therapy on autistic and adaptive behaviour outcomes. This supports the theoretical logic model of PACT therapy but also illuminates fundamental causal processes of social and adaptive development in autism over time: early social engagement in autism can be improved and this can have long-term generalised outcome effects.

Parental Conceptualizations of Autism and Deafness in British Deaf Children.

The co-occurrence of childhood deafness and autism raises complex challenges for diagnosis and family support. In this article, we explore with hearing and Deaf parents their observations of the interaction between deafness and autism and identify how the intersections of deafness and autism are conceptualized in everyday life. Eight parents participated (two of whom were Deaf BSL users) in semi-structured interviews in either BSL or spoken English. Data analysis was underpinned by a phenomenological approach in the hermeneutic tradition. Findings are discussed in terms of parents' perceptions of the relevance of deafness to their understanding of autism for their particular child, the effects of autism on sign and spoken language development and the relationship between deafness and autism in terms of their own and others' attributions of their children's characteristics. The significance of the findings for parental contributions' to diagnostic assessment and the tailoring of family support are considered.

Parents' Opinions about an Intervention to Manage Repetitive Behaviours in Young Children with Autism Spectrum Disorder: A Qualitative Study.

BACKGROUND: Early intervention for autism spectrum disorder (ASD) tends to focus on enhancing social communication skills. We report data collected via focus group discussions as part of a feasibility and acceptability pilot randomized controlled trial (RCT) about a new parent group intervention to manage restricted and repetitive behaviours (RRB) in young children with ASD. METHODS: The focus groups were led by two independent facilitators and followed a semi-structured topic guide with the aim of considering three key topics: experiences of participating in a RCT, opinions about the intervention and the impact of the intervention on the participants, their children and the family. RESULTS: Fourteen participants attended the focus groups. Most participants reported that they had little knowledge of RRB before attending the intervention and that it had had a positive impact on them, their children and their family. CONCLUSION: The findings support the view that there is an unmet need for a parent-mediated intervention focusing on RRB.

Parent-mediated social communication therapy for young children with autism (PACT): long-term follow-up of a randomised controlled trial.

BACKGROUND: It is not known whether early intervention can improve long-term autism symptom outcomes. We aimed to follow-up the Preschool Autism Communication Trial (PACT), to investigate whether the PACT intervention had a long-term effect on autism symptoms and continued effects on parent and child social interaction. METHODS: PACT was a randomised controlled trial of a parent-mediated social communication intervention for children aged 2-4 years with core autism. Follow-up ascertainment was done at three specialised clinical services centres in the UK (London, Manchester, and Newcastle) at a median of 5·75 years (IQR 5·42-5·92) from the original trial endpoint. The main blinded outcomes were the comparative severity score (CSS) from the Autism Diagnostic Observation Schedule (ADOS), the Dyadic Communication Assessment Measure (DCMA) of the proportion of child initiatiations when interacting with the parent, and an expressive-receptive language composite. All analyses followed the intention-to-treat principle. PACT is registered with the ISRCTN registry, number ISRCTN58133827. FINDINGS: 121 (80%) of the 152 trial participants (59 [77%] of 77 assigned to PACT intervention vs 62 [83%] of 75 assigned to treatment as usual) were traced and consented to be assessed between July, 2013, and September, 2014. Mean age at follow-up was 10·5 years (SD 0·8). Group difference in favour of the PACT intervention based on ADOS CSS of log-odds effect size (ES) was 0·64 (95% CI 0·07 to 1·20) at treatment endpoint and ES 0·70 (95% CI -0·05 to 1·47) at follow-up, giving an overall reduction in symptom severity over the course of the whole trial and follow-up period (ES 0·55, 95% CI 0·14 to 0·91, p=0·004). Group difference in DCMA child initiations at follow-up showed a Cohen's d ES of 0·29 (95% CI -0.02 to 0.57) and was significant over the course of the study (ES 0·33, 95% CI 0·11 to 0·57, p=0·004). There were no group differences in the language composite at follow-up (ES 0·15, 95% CI -0·23 to 0·53). INTERPRETATION: The results are the first to show long-term symptom reduction after a randomised controlled trial of early intervention in autism spectrum disorder. They support the clinical value of the PACT intervention and have implications for developmental theory. FUNDING: Medical Research Council.

Risk factors for eating disorder symptoms at 12 years of age: A 6-year longitudinal cohort study.

Eating disorders pose risks to health and wellbeing in young adolescents, but prospective studies of risk factors are scarce and this has impeded prevention efforts. This longitudinal study aimed to examine risk factors for eating disorder symptoms in a population-based birth cohort of young adolescents at 12 years. Participants from the Gateshead Millennium Study birth cohort (n = 516; 262 girls and 254 boys) completed self-report questionnaire measures of eating disorder symptoms and putative risk factors at age 7 years, 9 years and 12 years, including dietary restraint, depressive symptoms and body dissatisfaction. Body mass index (BMI) was also measured at each age. Within-time correlates of eating disorder symptoms at 12 years of age were greater body dissatisfaction for both sexes and, for girls only, higher depressive symptoms. For both sexes, higher eating disorder symptoms at 9 years old significantly predicted higher eating disorder symptoms at 12 years old. Dietary restraint at 7 years old predicted boys' eating disorder symptoms at age 12, but not girls'. Factors that did not predict eating disorder symptoms at 12 years of age were BMI (any age), girls' dietary restraint at 7 years and body dissatisfaction at 7 and 9 years of age for both sexes. In this population-based study, different patterns of predictors and correlates of eating disorder symptoms were found for girls and boys. Body dissatisfaction, a purported risk factor for eating disorder symptoms in young adolescents, developed concurrently with eating disorder symptoms rather than preceding them. However, restraint at age 7 and eating disorder symptoms at age 9 years did predict 12-year eating disorder symptoms. Overall, our findings suggest that efforts to prevent disordered eating might beneficially focus on preadolescent populations.

Neuropsychological study of IQ scores in offspring of parents with bipolar I disorder.

INTRODUCTION: Studies comparing IQ in Offspring of Bipolar Parents (OBP) with Offspring of Healthy Controls (OHC) have reported conflicting findings. They have included OBP with mental health/neurodevelopmental disorders and/or pharmacological treatment which could affect results. This UK study aimed to assess IQ in OBP with no mental health/neurodevelopmental disorder and assess the relationship of sociodemographic variables with IQ. METHODS: IQ data using the Wechsler Abbreviated Scale of Intelligence (WASI) from 24 OBP and 34 OHC from the North East of England was analysed using mixed-effects modelling. RESULTS: All participants had IQ in the average range. OBP differed statistically significantly from OHC on Full Scale IQ (p = .001), Performance IQ (PIQ) (p = .003) and Verbal IQ (VIQ) (p = .001) but not on the PIQ-VIQ split. OBP and OHC groups did not differ on socio-economic status (SES) and gender. SES made a statistically significant contribution to the variance of IQ scores (p = .001). CONCLUSIONS: Using a robust statistical model of analysis, the OBP with no current/past history of mental health/neurodevelopmental disorders had lower IQ scores compared to OHC. This finding should be borne in mind when assessing and recommending interventions for OBP.

Innovations in Practice: A case control and follow-up study of 'hard to reach' young people who suffered from multiple complex mental disorders.

BACKGROUND: Innovations Project (IP) was a new multidisciplinary team based within an inner city, walk-in health centre, North East England (throughout 2011). The aim was to describe the social and mental disorders of the hard to reach young people (HTRYP) from the IP and compare with a matched sample who attended a Community Mental Health Team (CMHT) and follow-up both samples 24 months after discharge. METHODS: A retrospective review of clinical case notes of YP who attended the IP and CMHT. A 24-month (postdischarge) follow-up evaluation of the mental state and social function of the YP in both groups using Health of the Nation Outcome Scales for Child and Adolescent Mental Health (HoNOSCA) and Children's Global Assessment Scale (CGAS). RESULTS: Thirty-six referrals were accepted over a one-year period by the IP, 31 met criteria for the HTRYP, 15 were offered individually tailored therapy. The HTRYP who were more deprived compared to the CMHT matched sample (n = 115), experienced a higher median number of mental disorders (n = 3 compared to CMHT n = 1), higher severity scores and lower levels of social function (HTRYP HoNOSCA mean: 19.1 (95% CI 15.9-22.2) and CMHT mean: 11.2 (95% CI 2.0-23.0) p = <.001, and HTRYP CGAS mean: 51.0 (95% CI 46.0-56.2) and CMHT mean: 58.9 (95% CI 52.9-64.8), p = .05). The HTRYP made significantly greater improvement compared to CMHTYP; (HoNOSCA p = <.001 and CGAS p = <.002) at discharge. A total of 13 HTRYP and 9 CMHT YP attended the follow-up review at 24 months. There was substantial variability in terms of social function between the YP within each sample. CONCLUSIONS: The term 'HTR' describes a state that may be often temporary, as opposed to lifelong. A bespoke service offering a developmental theoretical framework, regular reviews and an individualised care plan, was able to engage and had the potential to reduce morbidity suffered by HTRYP.

Treatment mechanism in the MRC preschool autism communication trial: implications for study design and parent-focussed therapy for children.

BACKGROUND: The PACT randomised-controlled trial evaluated a parent-mediated communication-focused treatment for children with autism, intended to reduce symptom severity as measured by a modified Autism Diagnostic Observation Schedule-Generic (ADOS-G) algorithm score. The therapy targeted parental behaviour, with no direct interaction between therapist and child. While nonsignificant group differences were found on ADOS-G score, significant group differences were found for both parent and child intermediate outcomes. This study aimed to better understand the mechanism by which the PACT treatment influenced changes in child behaviour though the targeted parent behaviour. METHODS: Mediation analysis was used to assess the direct and indirect effects of treatment via parent behaviour on child behaviour and via child behaviour on ADOS-G score. Alternative mediation was explored to study whether the treatment effect acted as hypothesised or via another plausible pathway. Mediation models typically assume no unobserved confounding between mediator and outcome and no measurement error in the mediator. We show how to better exploit the information often available within a trial to begin to address these issues, examining scope for instrumental variable and measurement error models. RESULTS: Estimates of mediation changed substantially when account was taken of the confounder effects of the baseline value of the mediator and of measurement error. Our best estimates that accounted for both suggested that the treatment effect on the ADOS-G score was very substantially mediated by parent synchrony and child initiations. CONCLUSIONS: The results highlighted the value of repeated measurement of mediators during trials. The theoretical model underlying the PACT treatment was supported. However, the substantial fall-off in treatment effect highlighted both the need for additional data and for additional target behaviours for therapy.

Cost-effectiveness analysis of a communication-focused therapy for pre-school children with autism: results from a randomised controlled trial.

BACKGROUND: Autism is associated with impairments that have life-time consequences for diagnosed individuals and a substantial impact on families. There is growing interest in early interventions for children with autism, yet despite the substantial economic burden, there is little evidence of the cost-effectiveness of such interventions with which to support resource allocation decisions. This study reports an economic evaluation of a parent-mediated, communication-focused therapy carried out within the Pre-School Autism Communication Trial (PACT). METHODS: 152 pre-school children with autism were randomly assigned to treatment as usual (TAU) or PACT + TAU. Primary outcome was severity of autism symptoms at 13-month follow-up. Economic data included health, education and social services, childcare, parental productivity losses and informal care. RESULTS: Clinically meaningful symptom improvement was evident for 53 % of PACT + TAU versus 41 % of TAU (odds ratio 1.91, p = 0.074). Service costs were significantly higher for PACT + TAU (mean difference £4,489, p < 0.001), but the difference in societal costs was smaller and non-significant (mean difference £1,385, p = 0.788) due to lower informal care rates for PACT + TAU. CONCLUSIONS: Improvements in outcome generated by PACT come at a cost. Although this cost is lower when burden on parents is included, the cost and effectiveness results presented do not support the cost-effectiveness of PACT + TAU compared to TAU alone. TRIAL REGISTRATION: Current Controlled Trials ISRCTN58133827.

Characteristics of young people with long term conditions close to transfer to adult health services.

BACKGROUND: For many young people with long term conditions (LTC), transferring from paediatric to adult health services can be difficult and outcomes are often reported to be poor. We report the characteristics and representativeness of three groups of young people with LTCs as they approach transfer to adult services: those with autism spectrum disorder with additional mental health problems (ASD); cerebral palsy (CP); or diabetes. METHODS: Young people aged 14 years-18 years 11 months with ASD, or those with diabetes were identified from children's services and those with CP from population databases. Questionnaires, completed by the young person and a parent, included the 'Mind the Gap' Scale, the Rotterdam Transition Profile, and the Warwick and Edinburgh Mental Wellbeing Scale. RESULTS: Three hundred seventy four young people joined the study; 118 with ASD, 106 with CP, and 150 with diabetes. Participants had a significant (p < 0.001) but not substantial difference in socio-economic status (less deprived) compared to those who declined to take part or did not respond. Condition-specific severity of participants was similar to that of population data. Satisfaction with services was good as the 'gap' scores (the difference between their ideal and current care) reported by parents and young people were small. Parents' satisfaction was significantly lower than their children's (p < 0.001). On every domain of the Rotterdam Transition Profile, except for education and employment, significant differences were found between the three groups. A larger proportion of young people with diabetes were in a more independent phase of participation than those with ASD or CP. The wellbeing scores of those with diabetes (median = 53, IQR: 47-58) and CP (median = 53, IQR: 48-60) were similar, and significantly higher than for those with ASD (median = 47, IQR: 41-52; p < 0.001). CONCLUSIONS: Having established that our sample of young people with one of three LTCs recruited close to transfer to adult services was representative, we have described aspects of their satisfaction with services, participation and wellbeing, noting similarities and differences by LTC. This information about levels of current functioning is important for subsequent evaluation of the impact of service features on the health and wellbeing of young people with LTCs following transfer from child services to adult services.

Individual common variants exert weak effects on the risk for autism spectrum disorders.

While it is apparent that rare variation can play an important role in the genetic architecture of autism spectrum disorders (ASDs), the contribution of common variation to the risk of developing ASD is less clear. To produce a more comprehensive picture, we report Stage 2 of the Autism Genome Project genome-wide association study, adding 1301 ASD families and bringing the total to 2705 families analysed (Stages 1 and 2). In addition to evaluating the association of individual single nucleotide polymorphisms (SNPs), we also sought evidence that common variants, en masse, might affect the risk. Despite genotyping over a million SNPs covering the genome, no single SNP shows significant association with ASD or selected phenotypes at a genome-wide level. The SNP that achieves the smallest P-value from secondary analyses is rs1718101. It falls in CNTNAP2, a gene previously implicated in susceptibility for ASD. This SNP also shows modest association with age of word/phrase acquisition in ASD subjects, of interest because features of language development are also associated with other variation in CNTNAP2. In contrast, allele scores derived from the transmission of common alleles to Stage 1 cases significantly predict case status in the independent Stage 2 sample. Despite being significant, the variance explained by these allele scores was small (Vm< 1%). Based on results from individual SNPs and their en masse effect on risk, as inferred from the allele score results, it is reasonable to conclude that common variants affect the risk for ASD but their individual effects are modest.

The lived experiences of relatives of autistic adults, and their perceptions of their relationships with autistic adults across multiple age-related transitions and demands: A qualitative interview study with reflexive thematic analysis.

BACKGROUND: There is a need to better understand autism across the life course, including the lives of both autistic people and supporting relatives. As part of a larger mixed methods cohort study involving autistic adults, carers and relatives this sub-study focused on the experiences of relatives alone to learn more about the lives of people from the wider personal networks. Our research questions were: 1. What are the experiences of family members who care for and/or support autistic adults, 2. How can the viewpoints of relatives add to what we know about transitions and challenges experienced by autistic adults, and 3. What strategies/support have been helpful for adults and relatives? METHODS: Relatives of autistic adults were purposively sampled and recruited using the Relatives/Carers cohort from the Adult Autism Spectrum Cohort-UK. 18 participants aged 31-81years who were related to 16 autistic adults aged 18-57years were interviewed for 24-91minutes. Interview transcripts were examined using reflexive thematic analysis. MAIN FINDINGS: Two overarching themes were developed, 'Family support goes a long way in caring for autistic adults' and 'When families turn to society for support' with subthemes. Relatives described benefits they had gained and their admiration for autistic adults. They reflected on how they gave support for independence in various contexts of dependence. They also identified the challenges that both autistic adults and families face navigating support systems (for example for healthcare and employment). An important novel outcome was the advocated value of role-models with lived experience who come from outside of the family. RECOMMENDATIONS: The findings lead to recommendations for: (i) Strategies to reduce the barriers for support that are faced by autistic individuals and relatives during crisis points; (ii) recognition and support for what enables both relatives and autistic adults to function independently (e.g. funded activities, flexible employment); (iii) future planning conversations to include relatives who can enhance knowledge and help plan for future care or support needs for autistic adults and (iv) opportunities for role models (persons with lived experience, autistic adults and relatives) to inspire others and disseminate knowledge. CONCLUSIONS: These findings add valuable insights into the experiences of relatives of autistic adults and challenge the reader to have greater appreciation of the many roles relatives can contribute across time and in a variety of contexts. These perspectives add important information for those working with and planning provision for autistic adults.

Generalisation of Social Communication Skills by Autistic Children During Play-Based Assessments Across Home, School and an Unfamiliar Research Setting.

We investigated autistic children's generalisation of social communication over time across three settings during a play-based assessment with different adults and explore the potential moderating effects on generalisation of age, nonverbal IQ and level of restricted and repetitive behaviours. The social communication abilities of 248 autistic children (2-11 years, 21% female, 22% single parent, 60% white) from three UK sites were assessed from 1984 video interactions in three contexts with three different interaction partners (parent/home, teaching assistant/school, researcher/clinic) at baseline, midpoint (+ 7m) and endpoint (+ 12m) within the Paediatric Autism Communication Trial-Generalised (PACT-G), a parent-mediated social communication intervention. Children's midpoint social communication at home generalised to school at midpoint and to clinic at endpoint. Generalisation was stronger from home to school and clinic than school to home and clinic. Generalisation was not moderated by age, nonverbal IQ or restricted and repetitive behaviour. Broader child development did not explain the pattern of results. The current study is the largest study to date to explore generalisation with autistic children and provides novel insight into their generalisation of social communication skills. Further research is needed to gain a more comprehensive understanding of facilitators of generalisation across settings and interaction partners in order to develop targeted strategies for interventions to enhance outcomes for young autistic children.

Diagnosing autism spectrum disorder: who will get a DSM-5 diagnosis?

BACKGROUND: Introduction of proposed criteria for DSM-5 Autism Spectrum Disorder (ASD) has raised concerns that some individuals currently meeting diagnostic criteria for Pervasive Developmental Disorder (PDD; DSM-IV-TR/ICD-10) will not qualify for a diagnosis under the proposed changes. To date, reports of sensitivity and specificity of the new criteria have been inconsistent across studies. No study has yet considered how changes at the 'sub domain' level might affect overall sensitivity and specificity, and few have included individuals of different ages and ability levels. METHODS: A set of DSM-5 ASD algorithms were developed using items from the Diagnostic Interview for Social and Communication Disorders (DISCO). The number of items required for each DSM-5 subdomain was defined either according to criteria specified by DSM-5 (Initial Algorithm), a statistical approach (Youden J Algorithm), or to minimise the number of false positives while maximising sensitivity (Modified Algorithm). The algorithms were designed, tested and compared in two independent samples (Sample 1, N = 82; Sample 2, N = 115), while sensitivity was assessed across age and ability levels in an additional dataset of individuals with an ICD-10 PDD diagnosis (Sample 3, N = 190). RESULTS: Sensitivity was highest in the Initial Algorithm, which had the poorest specificity. Although Youden J had excellent specificity, sensitivity was significantly lower than in the Modified Algorithm, which had both good sensitivity and specificity. Relaxing the domain A rules improved sensitivity of the Youden J Algorithm, but it remained less sensitive than the Modified Algorithm. Moreover, this was the only algorithm with variable sensitivity across age. All versions of the algorithm performed well across ability level. CONCLUSIONS: This study demonstrates that good levels of both sensitivity and specificity can be achieved for a diagnostic algorithm adhering to the DSM-5 criteria that is suitable across age and ability level.

Study protocol: longitudinal study of the transition of young people with complex health needs from child to adult health services.

BACKGROUND: Young people with complex health needs have impairments that can limit their ability to carry out day-to-day activities. As well as coping with other developmental transitions, these young people must negotiate the transfer of their clinical care from child to adult services. The process of transition may not be smooth and both health and social outcomes may suffer.Increasingly, policy-makers have recognised the need to ensure a smoother transition between children's and adult services, with processes that are holistic, individualised, and person-centred; however, there is little outcome data to support proposed models of care. This study aims to identify the features of transitional care that are potentially effective and efficient for young people with complex health needs making their transition. METHODS/DESIGN: Longitudinal cohort study. 450 young people aged 14 years to 18 years 11 months (with autism spectrum disorder and an additional mental health problem, cerebral palsy or diabetes) will be followed through their transition from child to adult services and will contribute data at baseline, 12, 24 and 36 months. We will collect data on: health and wellbeing outcomes (participation, quality of life, satisfaction with services, generic health status (EQ-5D-Y) and condition specific measure of disease control or management); exposure to proposed beneficial features of services (such as having a key worker, appropriate involvement of parents); socio-economic characteristics of the sample; use of condition-related health and personal social services; preferences for the characteristics of transitional care.We will us regression techniques to explore how outcomes vary by exposure to service features and by characteristics of the young people. These data will populate a decision-analytic model comparing the costs and benefits of potential alternative ways of organising transition services.In order to better understand mechanisms and aid interpretation, we will undertake qualitative work with 15 young people, including interviews, non-participant observation and diary collection. DISCUSSION: This study will evaluate the effect of service components of transitional care, rather than evaluation of specific models that may be unsustainable or not generalisable. It has been developed in response to numerous national and international calls for such evaluation.

Consensus statements on optimal adult post-autism diagnosis support and services: Delphi process following a UK survey of autistic adults, relatives and clinicians.

LAY ABSTRACT: Research has identified types of support helpful to autistic people, for example, physical and mental health interventions, psycho-education, peer support, developing positive identities and affiliation with social groups. However, accessing suitable post-autism diagnosis support and services is extremely difficult. We asked autistic adults, relatives and clinicians about their experiences of receiving and delivering post-autism diagnosis support/services. In Stage 1, 343 autistic adults and 45 relatives completed a survey. They answered questions about their experiences of UK autism post-diagnosis support/services for adults within 12 months after receiving a diagnosis. Thirty-five clinicians completed a similar survey. Just over half of adults and relatives said there was a follow-up appointment or discussion about support after diagnosis. Fewer than 40% received any support/services in 12 months after diagnosis. We used information from the surveys to create 11 statements describing characteristics of appropriate adult post-autism diagnosis support/services. In Stage 2, we asked clinicians for their views on the statements - they agreed with all of them. For example, those adults are offered an additional follow-up meeting after diagnosis and have access to mental and physical health services. We shared results with autistic adults, relatives and clinicians at two events. Some autistic adults, relatives and clinicians were positive about post-autism diagnosis support/services. However, they described many areas for improvement. The study findings can be used to define, develop and improve the types of adult post-diagnosis support services.

A Pilot Randomised Control Trial Exploring the Feasibility and Acceptability of Delivering a Personalised Modular Psychological Intervention for Anxiety Experienced by Autistic Adults: Personalised Anxiety Treatment-Autism (PAT-A).

Anxiety is commonly experienced by autistic people and impacts on quality of life and social participation. New anxiety interventions are required to effectively meet the needs of autistic people. Personalised Anxiety Treatment-Autism (PAT-A©) is a bespoke, modular approach to treating anxiety in up to 12 sessions. This study explored the feasibility and acceptability of delivering PAT-A© in the UK National Health Service (NHS). A single-blind randomised controlled trial design. Thirty-four autistic adults were recruited via clinical services and randomised to receive either PAT-A© or enhanced treatment as usual (CCSP). Outcome assessments relating to anxiety, quality of life and related constructs were completed at baseline, immediately post intervention; and at 3 and 12 months. Seventy-one percent of the PAT-A© group and 65% of the CCSP met diagnostic threshold for at least three anxiety disorders. Retention was good across both groups, with 82% (N = 14/17) completing the full course of PAT-A© and 71% (N = 12/17) attending both psychoeducational sessions in CCSP. 94% in PAT-A© and 82% in CCSP completed some follow up assessment 3 months post-intervention. Thematic analysis of interview data revealed that many participants valued the personalised approach, developed transferable skills and experienced positive changes to their anxiety. Participants were willing to be recruited and randomised, PAT-A© was feasible to deliver in the NHS and the trial methods and materials were acceptable. Our findings indicate that a fully powered clinical and cost-effectiveness trial of PAT-A© is warranted.

Comparison of Diagnostic Profiles of Deaf and Hearing Children with a Diagnosis of Autism.

There is limited research comparing the presentation of autism in deaf and hearing children and young people. These comparisons are important to facilitate accurate diagnosis, as rates of misdiagnosis and delay in diagnosis amongst deaf children and young people are high. The aim of this study was to compare diagnostic assessment profiles of a UK cohort of autistic deaf and hearing children and young people. The Autism Diagnostic Interview-Revised-Deaf adaptation was completed with the parents of 106 children and young people (deaf children = 65; hearing children = 41). The majority of items explored showed no significant differences between deaf and hearing children and young people. Differences were found in peer relationships, where autistic deaf participants were less likely to respond to the approaches of other children or play imaginatively with peers. These findings need to be taken into consideration by clinicians in the assessment process.

I-SOCIALISE: Results from a cluster randomised controlled trial investigating the social competence and isolation of children with autism taking part in LEGO® based therapy ('Play Brick Therapy') clubs in school environments.

LAY ABSTRACT: Autism is characterised by keen interests and differences in social interactions and communication. Activities that help autistic children and young people with social skills are commonly used in UK schools. LEGO® based therapy is a new activity that provides interesting and fun social opportunities for children and young people and involves building LEGO® models together. This study looked at LEGO® based therapy for the social skills of autistic children and young people in schools. It was a randomised controlled trial, meaning each school was randomly chosen (like flipping a coin) to either run LEGO® based therapy groups in school over 12 weeks and have usual support from school or other professionals, or only have usual support from school or other professionals. The effect of the LEGO® based therapy groups was measured by asking children and young people, their parents/guardians, and a teacher at school in both arms of the study to complete some questionnaires. The main objective was to see if the teacher's questionnaire answers about the children and young people's social skills changed between their first and second completions. The social skills of participants in the LEGO® based therapy groups were found to have improved in a small way when compared to usual support only. The study also found that LEGO® based therapy was not very costly for schools to run and parents/guardians and teachers said they thought it was good for their children and young people. We suggest further research into different potential benefits of LEGO® based therapy.

Play brick therapy to aid the social skills of children and young people with autism spectrum disorder: the I-SOCIALISE cluster RCT.

Background: Social skills interventions are commonly recommended to help children and young people with autism spectrum disorder develop social skills, but some struggle to engage in these interventions. LEGO® (LEGO System A/S, Billund, Denmark) based therapy, a group social skills intervention, aims to be more interesting and engaging. Objective: To evaluate the clinical effectiveness of LEGO® based therapy on the social and emotional skills of children and young people with autism spectrum disorder in school settings compared with usual support. Secondary objectives included evaluations of cost-effectiveness, acceptability and treatment fidelity. Design: A cluster randomised controlled trial randomly allocating participating schools to either LEGO® based therapy and usual support or usual support only. Setting: Mainstream schools in the north of England. Participants: Children and young people (aged 7-15 years) with autism spectrum disorder, their parent/guardian, an associated teacher/teaching assistant and a facilitator teacher/teaching assistant (intervention schools only). Intervention: Schools randomised to the intervention arm delivered 12 weekly sessions of LEGO® based therapy, which promotes collaborative play and encourages social problem-solving in groups of three children and young people with a facilitator (trained teacher or teaching assistant). Participants received usual support from school and community services. Participants in the control arm received usual support only. Research assistants and statisticians were blind to treatment allocation. Main outcome measure: The social skills subscale of the Social Skills Improvement System (SSIS), completed by the children and young people's unblinded teacher pre randomisation and 20 weeks post randomisation. The SSIS social skills subscale measures social skills including social communication, co-operation, empathy, assertion, responsibility and self-control. Participants completed a number of other pre- and post-randomisation measures of emotional health, quality of life, loneliness, problem behaviours, academic competence, service resource utilisation and adverse events. Results: A total of 250 children and young people from 98 schools were randomised: 127 to the intervention arm and 123 to the control arm. Intention-to-treat analysis of the main outcome measure showed a modest positive difference of 3.74 points (95% confidence interval -0.16 to 7.63 points, standardised effect size 0.18; p = 0.06) in favour of the intervention arm. Statistical significance increased in per-protocol analysis, with a modest positive difference (standardised effect size 0.21; p = 0.036). Cost-effectiveness of the intervention was found in reduced service use costs and a small increase in quality-adjusted life-years. Intervention fidelity and acceptability were positive. No intervention-related adverse events or effects were reported. Conclusions: The primary and pre-planned sensitivity analysis of the primary outcome consistently showed a positive clinical difference, with modest standardised effect sizes of between 0.15 and 0.21. There were positive health economics and qualitative findings, corroborated by the difference between arms for the majority of secondary outcomes, which were not statistically significant but favoured the intervention arm. Post hoc additional analysis was exploratory and was not used in drawing this conclusion. Given the small positive change, LEGO® based therapy for children and young people with autism spectrum disorder in schools should be considered. Limitations: The primary outcome measure was completed by an unblinded teacher (rather than by the facilitator). Future work: The study team recommends future research into LEGO® based therapy, particularly in school environments. Trial registration: This trial is registered as ISRCTN64852382. Funding: This award was funded by the National Institute for Health and Care Research (NIHR) Public Health Research programme (NIHR award ref: 15/49/32) and is published in full in Public Health Research; Vol. 11, No. 12. See the NIHR Funding and Awards website for further award information.

Autism spectrum disorder is characterised by difficulties with social relationships and communication, which can make it difficult to make friends. Social skills training is commonly used to help children and young people learn different social skills, but some children and young people do not enjoy these therapies. LEGO^® (LEGO System A/S, Billund, Denmark) based therapy takes a new approach by focusing on making the process interesting and fun. This research investigated the effect of LEGO^® based therapy groups in schools on the social and emotional abilities of children and young people with autism spectrum disorder. It was a randomised controlled trial, so each school that was taking part was randomly chosen to provide either usual support (from the school or NHS services) or 12 sessions of LEGO^® based therapy with a trained school staff member as well as usual support. Children and young people played one of three roles ­ the 'engineer' (gives instructions), the 'supplier' (finds the pieces) or the 'builder' (builds the model) ­ and worked together. Questionnaires completed by children and young people, their parents/guardians and teachers were used to look at the intervention's effects. The main objective was to see if there was a change in social skills measured by a teacher-completed questionnaire. Results showed that the social skills of children and young people in the LEGO^® based therapy groups did improve a little. We found that the intervention is not very costly for schools to run. Many parents/guardians and teachers thought that the intervention was beneficial and that the children and young people enjoyed it.