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.

Displaced Rohingya children at high risk for mental health problems: Findings from refugee camps within Bangladesh.

BACKGROUND: The 2017 political violence against the Rohingya people in the state of Rakhine resulted in a large influx of displaced populations into Bangladesh. Given harsh conditions and experiences in Myanmar, and the harrowing journey to the border, raised levels of child neurodevelopmental disorders (NDDs) and mental health problems were expected. METHODS: A team of child development professionals, physicians, psychologists, and developmental therapists screened 622 children in clinics within the refugee camps using the Developmental Screening Questionnaire (DSQ; 0-<2 years), and the Ten Questions Plus (TQP) for NDDs, and Strengths and Difficulties Questionnaire (SDQ; 2-16 years) for mental health problems. Any child positive on the DSQ or the TQP was assessed for NDDs. RESULTS: Only 4.8% children aged 0-<2 years and 7.3% children aged >2-16 years screened positive for NDDs, comparable with a local Bangladesh population. However, 52% of children were in the abnormal range for emotional symptoms on the SDQ, and 25% abnormal for peer problems. Significant risk factors were being parentless and having lost one or more family members in the recent crisis. CONCLUSIONS: This screening study provides objective evidence of the urgent need for psychosocial support of Rohingya children within camps, with special attention to those without parents, including monitoring of their well-being and counselling of families and other care providers.

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.

Measuring self-reported quality of life in 8- to 11-year-old children born with gastroschisis: Is the KIDSCREEN questionnaire acceptable?

BACKGROUND: Children born with gastroschisis have a good prognosis but require surgical correction and long-term follow up. There has been little research on the impact of gastroschisis on the child's health-related quality of life (QoL). The aim was to assess face and content validity of the KIDSCREEN-52 questionnaire as a measurement of self- and proxy-reported QoL in children born with gastroschisis and to evaluate self-reported QoL in these children compared with the reference population. METHODS: In this cross-sectional exploratory study, we used the validated KIDSCREEN-52 questionnaire and individual interview with 8- to 11-year-old children born with gastroschisis who were identified from the Northern Congenital Abnormality Survey. Self-reported QoL scores were compared with age-matched UK norms by using the two-sample t test. RESULTS: Ten children (median age 9.6 years, interquartile range 8.3-11.0) and their parents participated. Children found KIDSCREEN a helpful tool to explore their feelings and that it covered life aspects important to them. Parents believed that all priority areas were represented and that it was straightforward for their children to complete. In nine KIDSCREEN domains, children with gastroschisis had similar QoL scores to those in the reference population, and in one (psychological well-being) the mean score was significantly better (p = 0.03). All children described their health as good/very good or excellent; eight said they would not like to change anything about their body. CONCLUSION: The KIDSCREEN questionnaire has adequate face and content validity as a measure of QoL in children with gastroschisis and is acceptable to both children and parents.

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.

Prevalence and correlates of use of complementary and alternative medicine in children with autism spectrum disorder in Europe.

UNLABELLED: This study examined the prevalence and correlates of use of complementary and alternative medicine (CAM) among a sample of children with autism spectrum disorder (ASD) < 7 years in 18 European countries (N = 1,680). Forty-seven percent of parents reported having tried any CAM approach in the past 6 months. Diets and supplements were used by 25 % of the sample and mind-body practices by 24 %; other unconventional approaches were used by 25 % of the families, and a minority of parents reported having tried any invasive or potentially harmful approach (2 %). Parents in Eastern Europe reported significantly higher rates of CAM use. In the total sample, children with lower verbal ability and children using prescribed medications were more likely to be receiving diets or supplements. Concurrent use of high levels of conventional psychosocial intervention was significantly associated with use of mind-body practices. Higher parental educational level also increased the likelihood of both use of diets and supplements and use of mind-body practices. CONCLUSION: The high prevalence of CAM use among a sample of young children with ASD is an indication that parents need to be supported in the choice of treatments early on in the assessment process, particularly in some parts of Europe. WHAT IS KNOWN: • Use of complementary and alternative medicine (CAM) in children with autism spectrum disorder is common. • In non-EU samples, parents with higher educational level and parents of low functioning children are more likely to use CAM with their children. What is New: • This study provides the first data on prevalence and correlates of use of CAM approaches in a large sample of young children with autism in Europe (N = 1,680). • Rates of CAM use were particularly high in Eastern Europe and correlates of use varied by type of CAM across Europe.

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.

Validation of a rapid neurodevelopmental assessment tool for 5 to 9 year-old children in Bangladesh.

OBJECTIVE: To validate a rapid neurodevelopmental assessment tool for use by child care professionals to determine neurodevelopmental impairments (NDIs) in children ages 5-9 years (61-108 months) in Bangladesh. STUDY DESIGN: In a convenience sample of community children (n = 18), interrater reliability was determined between 6 testers. Validity was determined in 121 children by simultaneous administration of a test of adaptive behavior (AB) (ie, the Independent Behavior Assessment Scale, or Gold Standard I) and IQ tests (Wechsler Preschool and Primary Scales of Intelligence or the Wechsler Intelligence Scales for Children, or Gold Standard II) by child psychologists. RESULTS: Interrater reliability was excellent (kappa = 1.00). Significantly lower scores were obtained on AB and IQ tests in those identified with "any (≥1) NDI" and most specific NDIs. Sensitivity and specificity for "any NDI" with (a) "significant difficulties" (defined as AB z-scores < -2 SD and/or IQ < 70) or (b) "mild difficulties included" (AB z scores < -1 SD and/or IQ < 85) were 84% and 57%; and 83% and 70%, respectively. CONCLUSION: The rapid neurodevelopmental assessment tool shows promise as a tool for use by a range of professionals for identifying NDIs in children of primary school age. Further refinement for identifying specific impairments is needed.

Interventions based on the Theory of Mind cognitive model for autism spectrum disorder (ASD).

BACKGROUND: The 'Theory of Mind' (ToM) model suggests that people with autism spectrum disorder (ASD) have a profound difficulty understanding the minds of other people - their emotions, feelings, beliefs, and thoughts. As an explanation for some of the characteristic social and communication behaviours of people with ASD, this model has had a significant influence on research and practice. It implies that successful interventions to teach ToM could, in turn, have far-reaching effects on behaviours and outcome. OBJECTIVES: To review the efficacy of interventions based on the ToM model for individuals with ASD. SEARCH METHODS: In August 2013 we searched CENTRAL, Ovid MEDLINE, Embase, CINAHL, PsycINFO, ERIC, Social Services Abstracts, AutismData, and two trials registers. We also searched the reference lists of relevant papers, contacted authors who work in this field, and handsearched a number of journals. SELECTION CRITERIA: Review studies were selected on the basis that they reported on an applicable intervention (linked to ToM in one of four clearly-defined ways), presented new randomised controlled trial data, and participants had a confirmed diagnosis of an autism spectrum disorder. Studies were selected by two review authors independently and a third author arbitrated when necessary. DATA COLLECTION AND ANALYSIS: Risk of bias was evaluated and data were extracted by two review authors independently; a third author arbitrated when necessary. Most studies were not eligible for meta-analysis, the principal reason being mis-matching methodologies and outcome measures. Three small meta-analyses were carried out. MAIN RESULTS: Twenty-two randomised trials were included in the review (N = 695). Studies were highly variable in their country of origin, sample size, participant age, intervention delivery type, and outcome measures. Risk of bias was variable across categories. There were very few studies for which there was adequate blinding of participants and personnel, and some were also judged at high risk of bias in blinding of outcome assessors. There was also evidence of some bias in sequence generation and allocation concealment. Not all studies reported data that fell within the pre-defined primary outcome categories for the review, instead many studies reported measures which were intervention-specific (e.g. emotion recognition). The wide range of measures used within each outcome category and the mixed results from these measures introduced further complexity when interpreting results.Studies were grouped into four main categories according to intervention target/primary outcome measure. These were: emotion recognition studies, joint attention and social communication studies, imitation studies, and studies teaching ToM itself. Within the first two of these categories, a sub-set of studies were deemed suitable for meta-analysis for a limited number of key outcomes.There was very low quality evidence of a positive effect on measures of communication based on individual results from three studies. There was low quality evidence from 11 studies reporting mixed results of interventions on measures of social interaction, very low quality evidence from four studies reporting mixed results on measures of general communication, and very low quality evidence from four studies reporting mixed results on measures of ToM ability.  The meta-analysis results we were able to generate showed that interventions targeting emotion recognition across age groups and working with people within the average range of intellectual ability had a positive effect on the target skill, measured by a test using photographs of faces (mean increase of 0.75 points, 95% confidence interval (CI) 0.22 to 1.29 points, Z = 2.75, P < 0.006, four studies, N = 105). Therapist-led joint attention interventions can promote production of more joint attention behaviours within adult-child interaction (mean increase of 0.55 points, 95% CI 0.11 to 0.99 points, Z = 2.45, P value = 0.01, two studies, N = 88). Further analysis undermines this conclusion somewhat by demonstrating that there was no clear evidence that intervention can have an effect on joint attention initiations as measured using a standardised assessment tool (mean increase of 0.23 points, 95% CI -0.48 to 0.94 points, Z = 0.63, P value = 0.53, three studies, N = 92). No adverse effects were apparent. AUTHORS' CONCLUSIONS: While there is some evidence that ToM, or a precursor skill, can be taught to people with ASD, there is little evidence of maintenance of that skill, generalisation to other settings, or developmental effects on related skills. Furthermore, inconsistency in findings and measurement means that evidence has been graded of 'very low' or 'low' quality and we cannot be confident that suggestions of positive effects will be sustained as high-quality evidence accumulates. Further longitudinal designs and larger samples are needed to help elucidate both the efficacy of ToM-linked interventions and the explanatory value of the ToM model itself. It is possible that the continuing refinement of the ToM model will lead to better interventions which have a greater impact on development than those investigated to date.

Treatment mechanism of the WHO caregiver skills training intervention for autism delivered in community settings.

This study examined the mechanism of effect of the WHO Caregiver Skills Training (CST) through secondary analysis of a pilot RCT conducted in community settings. Participants were 86 caregivers (77% mothers) of children with ASD (78% male, mean age: 44.8 months) randomized to CST (n = 43) or treatment as usual (n = 43). The primary outcomes, measured at baseline (t1), immediately post-intervention (t2), and 3 months post-intervention (t3), were derived from the coding of caregiver-child free play interactions with the Brief Observation of Social-Communication Change (BOSCC) and the Joint Engagement Rating Inventory scale (JERI). At t3 positive treatment main effects had been observed for caregiver skills supportive of the interaction and for flow of the interaction (JERI), albeit only non-significant changes in the expected direction for child outcomes: autism phenotypic behaviors (BOSCC), joint engagement and availability to interact (JERI). This study tested the theory of change of CST, hypothesizing that the intervention would lead to an improvement on all child and dyad outcomes through an increase in the caregiver skills supportive of the interaction. Serial mediation analyses revealed that the effect of the intervention was significantly influenced by change in caregiver skills. Participation in the intervention led to notable increases in caregiver skills at t2 and t3, which subsequently contributed to improvements at t3 in flow of the interaction, autism phenotypic behavior, joint engagement, and availability to interact. We confirmed our a priori hypothesis showing that change in caregiver skills significantly mediated the effect of treatment on the dyad primary outcome, as well as on the other child outcomes that had shown non-significant changes in the expected direction. Implications for intervention design and policy making in the context of public health services are discussed.

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.

Parent-mediated early intervention for young children with autism spectrum disorders (ASD).

BACKGROUND: Young children with autism spectrum disorders (ASD) have impairments in the areas of communication and social interaction and often display repetitive or non-compliant behaviour. This early pattern of difficulties is a challenge for parents. Therefore, approaches that help parents develop strategies for interaction and management of behaviour are an obvious route for early intervention in ASD. This review updates a Cochrane review first published in 2002 but is based on a new protocol. OBJECTIVES: To assess the effectiveness of parent-mediated early interventions in terms of the benefits for both children with ASD and their parents and to explore some potential moderators of treatment effect. SEARCH METHODS: We searched a range of psychological, educational and biomedical databases including CENTRAL, MEDLINE, Embase, PsycINFO and ERIC in August 2012. As this is an update of a previous review, we limited the search to the period following the original searches in 2002. Bibliographies and reference lists of key articles were searched, field experts were contacted and key journals were handsearched. SELECTION CRITERIA: We included only randomised controlled trials of early intervention for children with ASD. The interventions in the experimental condition were mediated by parents; the control conditions included no treatment, treatment as usual, waiting list, alternative child-centred intervention not mediated by parents, or alternative parent-mediated intervention of hypothesised lesser effect than the experimental condition. DATA COLLECTION AND ANALYSIS: Two review authors (HM and IPO) independently screened articles identified in the search and decided which articles should be retrieved in full. For each included study, two review authors (IPO and EH) extracted and recorded data, using a piloted data collection form. Two review authors (IPO and HM) assessed the risk of bias in each study. We performed data synthesis and analysis using The Cochrane Collaboration's Review Manager 5.1 software. MAIN RESULTS: The review includes 17 studies from six countries (USA, UK, Australia, Canada, Thailand and China), which recruited 919 children with ASD. Not all 17 studies could be compared directly or combined in meta-analyses due to differences in the theoretical basis underpinning interventions, the duration and intensity of interventions, and the outcome measurement tools used. Data from subsets of 10 studies that evaluated interventions to enhance parent interaction style and thereby facilitate children's communication were included in meta-analyses. The largest meta-analysis combined data from 316 participants in six studies and the smallest combined data from 55 participants in two studies. Findings from the remaining seven studies were reported narratively.High risk of bias was evident in the studies in relation to allocation concealment and incomplete outcome data; blinding of participants was not possible.Overall, we did not find statistical evidence of gains from parent-mediated approaches in most of the primary outcomes assessed (most aspects of language and communication - whether directly assessed or reported; frequency of child initiations in observed parent-child interaction; child adaptive behaviour; parents' stress), with findings largely inconclusive and inconsistent across studies. However, the evidence for positive change in patterns of parent-child interaction was strong and statistically significant (shared attention: standardised mean difference (SMD) 0.41; 95% confidence interval (CI) 0.14 to 0.68, P value < 0.05; parent synchrony: SMD 0.90; 95% CI 0.56 to 1.23, P value < 0.05). Furthermore, there is some evidence suggestive of improvement in child language comprehension, reported by parents (vocabulary comprehension: mean difference (MD 36.26; 95% CI 1.31 to 71.20, P value < 0.05). In addition, there was evidence suggesting a reduction in the severity of children's autism characteristics (SMD -0.30, 95% CI -0.52 to -0.08, P value < 0.05). However, this evidence of change in children's skills and difficulties as a consequence of parent-mediated intervention is uncertain, with small effect sizes and wide CIs, and the conclusions are likely to change with future publication of high-quality RCTs. AUTHORS' CONCLUSIONS: The review finds some evidence for the effectiveness of parent-mediated interventions, most particularly in proximal indicators within parent-child interaction, but also in more distal indicators of child language comprehension and reduction in autism severity. Evidence of whether such interventions may reduce parent stress is inconclusive. The review reinforces the need for attention to be given to early intervention service models that enable parents to contribute skilfully to the treatment of their child with autism. However, practitioners supporting parent-mediated intervention require to monitor levels of parent stress. The ability to draw conclusions from studies would be improved by researchers adopting a common set of outcome measures as the quality of the current evidence is low.

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.

Attenuation of change blindness in children with autism spectrum disorders.

Change blindness refers to the difficulty most people find in detecting a difference between two pictures when these are presented successively, with a brief interruption between. Attention at the site of the change is required for detection. A number of studies have investigated change blindness in adults and children with autism spectrum disorders (ASD). Some have produced evidence that people with ASD find changes to social stimuli harder to detect and changes to non-social stimuli easier to detect, relative to comparison participants. However, other studies have produced entirely contradictory findings. There is a need for consistency in methodology to aid understanding of change blindness and attentional processes in ASD. Here, we replicate a change blindness study previously carried out with typically developing (TD) children and adults and with adults with ASD. Results reveal attenuated change blindness for non-social stimuli in children with ASD relative to TD norms. Our results are interpreted, alongside others' findings, as potentially indicative of a complex relationship between different influences on attention over time.

"We couldn't think in the box if we tried. We can't even find the damn box": A qualitative study of the lived experiences of autistic adults and relatives of autistic adults.

Autistic children grow to become autistic adults, and autism is increasingly diagnosed in adulthood and later life. This qualitative study aimed to understand experiences of autism throughout adulthood. A national cohort study of autistic adults and relatives of autistic adults (ASC-UK), enabled purposive recruitment of a diverse sample. Semi-structured interviews were conducted with 29 autistic adults (aged 20-71 years), mostly diagnosed in adulthood, and 16 relatives (aged 31-81 years) of autistic adults diagnosed across both childhood and adulthood (including some with learning disability). Interview topics included health, relationships, education, employment, quality of life and everyday experiences. Thematic analysis of the accounts of the autistic adults identified six key themes relating to their experiences: (1) diagnosis as validating yet limiting; (2) supportive and non-supportive social agents; (3) the "invisibility" of the needs of autistic adults; (4) health in the context of autism; (5) staying 'outside' the circle; and (6) multiple lives with autism. Data from relatives about autistic adult experiences gave additional perspectives on these themes. Experiences reported in other studies-of 'difference' from others, challenges of social engagement, and learning to 'conform' to society's expectations-were evident and relevant to male and female autistic adults, across all age groups, and unrelated to stage of life when diagnosed. Some expressed disappointment with their lives, but others were proud of their achievements. Education and employment, whilst challenging for many, were also rewarding for some. Health care and social services were often experienced as inaccessible, inappropriate, or lacking understanding of the individual's needs. We conclude that greater public understanding of autism as experienced in adulthood is needed. Key priorities are improving the availability of 'appropriate' health and social care services for autistic adults and families, and providing practical support to enable enhanced participation in life.

Pilot Randomized Controlled Trial of the WHO Caregiver Skills Training in Public Health Services in Italy.

Parents of children with ASD (N = 86; mean age 44.8 months; 67 boys) were randomized to either WHO Caregiver Skills Training (CST) delivered in public health settings in Italy or enhanced treatment-as-usual. Primary blinded outcomes were 3-months post-intervention change scores of autism severity and engagement during caregiver-child interaction. CST was highly acceptable to caregivers and feasibly delivered by trained local clinicians. Intention-to-treat analysis showed a large and significant effect on parent skills supporting joint engagement and a smaller significant effect on flow of interaction. Expected changes in child autism severity and joint engagement did not meet statistical significance. Analysis of secondary outcomes showed a significant effect on parenting stress, self-efficacy, and child gestures. Strategies to improve the effectiveness of CST are discussed.

Adapting and validating the Autism Diagnostic Observation Schedule Version 2 for use with deaf children and young people.

We report a Delphi Consensus modification and first validation study of the Autism Diagnostic Observation Schedule - 2 with deaf children and young people (ADOS-2 Deaf adaptation). Validation included 122 deaf participants (aged 2-18 years), 63 with an Autism Spectrum Disorder (ASD). This was compared to a National Institute for Health and Clinical Excellence (NICE) guideline standard clinical assessment by blinded independent specialist clinicians. Results showed overall sensitivity 73% (95%CI 60%, 83%); specificity 71% (95%CI 58%, 82%), and for the more common modules 1-3 (combined as in previous studies) sensitivity 79% (95% CI 65-89%); specificity 79% (95% CI 66-89%) suggesting this instrument will be a helpful addition for use with deaf children and young people.

Developing the FEEDS toolkit of parent-delivered interventions for eating, drinking and swallowing difficulties in young children with neurodisability: findings from a Delphi survey and stakeholder consultation workshops.

BACKGROUND: Young children with neurodisability commonly experience eating, drinking and swallowing difficulties (EDSD). Little is documented about which interventions and outcomes are most appropriate for such children. We aimed to seek consensus between parents of children with neurodisability and health professionals on the appropriate interventions and outcomes to inform future clinical developments and research studies. METHODS: Two populations were sampled: parents of children aged up to 12 years with neurodisability who experienced EDSD; health professionals working with children and young people (aged 0-18 years) with neurodisability with experience of EDSD. Participants had taken part in a previous national survey and were invited to take part in a Delphi survey and/or consultation workshops. Two rounds of this Delphi survey sought agreement on the appropriate interventions and outcomes for use with children with neurodisability and EDSD. Two stakeholder consultation workshops were iterative, with the findings of the first discussed at the second, and conclusions reached. RESULTS: A total of 105 parents and 105 health professionals took part. Parents and health professionals viewed 19 interventions and 10 outcomes as essential. Interventions related to improvement in the physical aspects of a child's EDSD, behavioural changes of the child or parent, and changes in the child or family's well-being. Both parents and health professionals supported a 'toolkit' of interventions that they could use together in shared decision making to prioritise and implement timely interventions appropriate to the child. CONCLUSIONS: This study identified interventions viewed as essential to consider for improving EDSD in children with neurodisability. It also identified several key outcomes that are valued by parents and health professionals. The Focus on Early Eating, Drinking and Swallowing (FEEDS) Toolkit of interventions to improve EDSD in children with neurodisability has been developed and now requires evaluation regarding its use and effectiveness.

Understanding the heterogeneity of anxiety in autistic youth: A person-centered approach.

The present study aimed to examine anxiety profiles among children and adolescents on the autism spectrum. It further aimed to characterize the association between the identified anxiety profiles and key clinical and developmental variables. The Spence Children's Anxiety Scale-Parent Version (SCAS-P) data from a large international pooled sample of 870 caregivers of autistic children and adolescents (Mage  = 11.6 years, SDage  = 2.77; 107 females) was used. Latent profile analysis identified a three-anxiety profile solution exhibiting high entropy (0.80) and high latent profile probabilities, with good classification accuracy. Identified profiles fell along the severity spectrum and were named as the mild (n = 498), moderate (n = 272) and severe (n = 100) anxiety profiles. There were no statistically significant differences between the three anxiety profiles in terms of sex distribution. Participants in the mild profile were significantly younger than those in the severe profile, had significantly fewer social communication difficulties than youth in the moderate anxiety profile group and had significantly fewer restricted and repetitive behaviors and lower cognitive functioning scores compared to participants in moderate and severe anxiety profiles. This is the first study to move beyond identifying associations and group-level differences to exploring and identifying characteristics of anxiety-based subgroups at an individual level that differ on key clinical and developmental variables. The subgroups identified in this study are a preliminary, yet important, first step towards informing future assessment and individualized interventions aiming to support young people on the autism spectrum to reduce and manage anxiety. LAY SUMMARY: This study tried to understand if there are subgroups of autistic young people who may have similar anxiety profiles. We found that we could meaningfully group young people into three groups based on how severe the anxiety symptoms their caregivers reported were: a group with low levels of anxiety, those with moderate anxiety, and those with more severe anxiety. We also found that the young people in the mild group were younger, had fewer autism traits and lower levels of intellectual functioning than young people in the other two groups.