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.