Annual Research Review: Achieving universal health coverage for young children with autism spectrum disorder in low- and middle-income countries: a review of reviews.

BACKGROUND: Autism presents with similar prevalence and core impairments in diverse populations. We conducted a scoping review of reviews to determine key barriers and innovative strategies which can contribute to attaining universal health coverage (UHC), from early detection to effective interventions for autism in low- and middle-income countries (LAMIC). METHODS: A systematic literature search of review articles was conducted. Reviews relevant to the study research question were included if they incorporated papers from LAMIC and focused on children (

Developmental trajectories in infants and pre-school children with Neurofibromatosis 1.

BACKGROUND: Children with Neurofibromatosis 1 (NF1) show cognitive, behavioural and social differences compared to their peers. However, the age and sequence at which these differences begin to emerge is not fully understood. This prospective cohort study examines the cognitive, behavioural, ADHD trait and autism symptom development in infant and pre-school children with NF1 compared with typically developing (TD) children without a family history of neurodevelopmental conditions. METHODS: Data from standardised tests was gathered at 5, 10, 14, 24 and 36 months of age (NF1 n = 35, TD n = 29). Developmental trajectories of cognitive (Mullen Scales of Early Learning, MSEL) and adaptive behavioural (Vineland Adaptive Behavior Scales, VABS) development from 5 to 36 months were analysed using linear mixed modelling. Measures of ADHD (Child Behavior Checklist) and autism traits (ADOS-2, BOSA-MV and ADI-R) were assessed at 24 and 36 months. RESULTS: The developmental trajectory of cognitive skills (all domains of the MSEL) and behavioural skills (four domains of the VABS) differed significantly between NF1 and TD groups. Post-hoc tests demonstrated that the NF1 participants scored significantly lower than TD participants at 24 months on all MSEL and VABS domains. The NF1 cohort demonstrated higher mean autism and ADHD traits at 24 months and 14% of the NF1 cohort met a research diagnostic classification for autism at 36 months. LIMITATIONS: The study has a relatively small sample size due to variable retention and rolling recruitment. Due to limitations imposed by the COVID-19 pandemic, we utilised the Brief Observation of Symptoms of Autism for Minimally Verbal children (BOSA-MV) for some participants, which was administered online and may not gather as accurate a picture of traits as ADOS-2. The BOSA-MV was utilised for 41% of participants with NF1 at 36 months compared to 11% at 24 months. This may explain the reduction in the percentage of children with NF1 that met autism criteria at 36 months. CONCLUSIONS: By 24 months of age, the NF1 cohort show lower cognitive skills and adaptive behaviour and higher levels of autism and ADHD traits as compared to TD children. This has implications for developmental monitoring and referral for early interventions. TRIAL REGISTRATION: Not applicable.

The development, feasibility and acceptability of Empower-Autism: A new psychoeducational and psychotherapeutic programme for caregivers of children recently diagnosed with autism.

LAY ABSTRACT: What is already known about the topic?Parents and carers face many challenges following their child's autism diagnosis. They often look for information, and social and emotional support. There has been relatively little research into how best to provide this support and this means that there is no evidence to guide the delivery of services. Studies have suggested that an approach called Acceptance and Commitment Therapy can help parents and carers with their adjustment and emotional wellbeing.What does this article add?This article describes the development of a new group-based programme to address the diverse needs of caregivers after their child's autism diagnosis. The new programme was developed with caregivers, autistic people and professionals. It was called Empower-Autism and contained lots of information about autism and strategies to support autistic children, alongside therapeutic aspects based on Acceptance and Commitment Therapy. The programme was delivered to 29 parents/carers in three groups. Attendance at the groups was satisfactory. Both parents/carers and facilitators liked and valued the programme and found it accessible. They made suggestions for improvements. After the programme, parents and carers described improved wellbeing. They felt more positive and more connected to other people. They also described parenting their child in a more informed and sensitive way.Implications for practice, research or policyThe new programme is being tested within a large clinical trial. If there are positive results, the programme could be recommended for delivery and this would address an important gap in evidence-based practice.

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.

Using implementation science frameworks to explore barriers and facilitators for parents' use of therapeutic strategies following a parent-mediated autism intervention.

LAY ABSTRACT: Many early autism interventions teach parents therapeutic strategies to help them adjust their communication style with their children. Research has shown that this behaviour change in parents leads to improvements in child communication. It is, therefore, important to learn what factors support or hinder parents in their use of therapeutic strategies learned in such interventions. This study set out to interview parents who had participated in a research trial of the Paediatric Autism Communication Therapy-Generalised intervention. We interviewed 27 caregivers and explored their use of the strategies up to 2 years after the end of the research trial. Qualitative frameworks were used to inform interview questions and data analysis. These frameworks focused on a range of contextual factors, including parents' characteristics, their context and features of the intervention. Parents reported barriers and facilitators to using Paediatric Autism Communication Therapy-Generalised strategies across three themes: Motivating Factors; Opportunity and Support; Parent Characteristics. One of these themes, Motivating Factors, was further divided into the subthemes Compatibility and Buy-In and Alignment of Goals and Outcomes. Almost all parents reported continued use of the Paediatric Autism Communication Therapy-Generalised strategies. Facilitators included parental confidence in using the strategies and barriers included child's behaviour. Consideration of these factors can inform ways to better support parents in future autism interventions.

REACH-ASD: a UK randomised controlled trial of a new post-diagnostic psycho-education and acceptance and commitment therapy programme against treatment-as-usual for improving the mental health and adjustment of caregivers of children recently diagnosed with autism spectrum disorder.

BACKGROUND: Autism is a neurodevelopmental disability affecting over 1% of UK children. The period following a child's autism diagnosis can present real challenges in adaptation for families. Twenty to 50% of caregivers show clinically significant levels of mental health need within the post-diagnostic period and on an ongoing basis. Best practice guidelines recommend timely post-diagnostic family support. Current provision is patchy, largely unevidenced, and a source of dissatisfaction for both families and professionals. There is a pressing need for an evidenced programme of post-diagnostic support focusing on caregiver mental health and adjustment, alongside autism psycho-education. This trial tests the clinical and cost-effectiveness of a new brief manualised psychosocial intervention designed to address this gap. METHODS: This is a multi-centre two-parallel-group single (researcher)-blinded randomised controlled trial of the Empower-Autism programme plus treatment-as-usual versus usual local post-diagnostic offer plus treatment-as-usual. Caregivers of children aged 2-15 years with a recent autism diagnosis will be recruited from North West England NHS or local authority centres. Randomisation is individually by child, with one "index" caregiver per child, stratified by centre, using 2:1 randomisation ratio to assist recruitment and timely intervention. Empower-Autism is a group-based, manualised, post-diagnostic programme that combines autism psycho-education and psychotherapeutic components based on Acceptance and Commitment Therapy to support caregiver mental health, stress management and adjustment to their child's diagnosis. The comparator is any usual local group-based post-diagnostic psycho-education offer. Receipt of services will be specified through health economic data. PRIMARY OUTCOME: caregiver mental health (General Health Questionnaire-30) at 52-week follow-up. SECONDARY OUTCOMES: key caregiver measures (wellbeing, self-efficacy, adjustment, autism knowledge) at 12-, 26- and 52-week follow-up and family and child outcomes (wellbeing and functioning) at 52-week endpoint. SAMPLE: N=380 (approximately 253 intervention/127 treatment-as-usual). Primary analysis will follow intention-to-treat principles using linear mixed models with random intercepts for group membership and repeated measures. Cost-effectiveness acceptability analyses will be over 52 weeks, with decision modelling to extrapolate to longer time periods. DISCUSSION: If effective, this new approach will fill a key gap in the provision of evidence-based care pathways for autistic children and their families. TRIAL REGISTRATION: ISRCTN 45412843 . Prospectively registered on 11 September 2019.

Combined social communication therapy at home and in education for young autistic children in England (PACT-G): a parallel, single-blind, randomised controlled trial.

BACKGROUND: Autistic children can have difficulty generalising treatment effects beyond the immediate treatment context. Paediatric Autism Communication Therapy (PACT) has been successful when delivered in the clinic. Here we tested the Paediatric Autism Communication Therapy-Generalised (PACT-G) intervention combined between home and education settings for its overall effect and mechanistic transmission of effect across contexts. METHODS: In this parallel, single-blind, randomised, controlled trial, we recruited autistic children aged 2-11 years in urban or semi-urban areas in Manchester, Newcastle, and London, England. Children needed to meet core autism criteria on Autism Diagnostic Observation Schedule-second edition (ADOS-2) and parent-rated Social Communication Questionnaire (SCQ-lifetime), and children older than 5 years were included if they had intentional communication but expressive language equivalent of age 4 years or younger. Eligible children were randomly assigned (1:1), using block randomisation (random block sizes of 2 and 4) and stratified for site, age (2-4 years vs 5-11 years), and gender, to either PACT-G plus treatment as usual or treatment as usual alone. Research assessors were masked to treatment allocation. The PACT-G intervention was delivered by a therapist in parallel to the child's parents at home and to learning-support assistants (LSA) at their place of education, using both in-person and remote sessions over a 6 month period, to optimise adult-child social interaction. Treatment as usual included any health support or intervention from education or local community services. The primary outcome was autism symptom severity using the ADOS-2, as measured by researchers, at 12 months versus baseline. Secondary outcomes were Brief Observation of Social Communication Change (BOSCC) and dyadic social interaction between child and adult across contexts, both at 12 months. Other secondary outcome measures were assessed using the following composites: language, anxiety, repetitive behaviour, adaptive behaviour, parental wellbeing, child health-related quality of life, and disruptive behaviour. Assessments were done at baseline, 7 months, and 12 months. We used an intention-to-treat (ITT) analysis of covariance for the efficacy outcome measures. Adverse events were assessed by researchers for all trial families at each contact and by therapists in the PACT-G group at each visit. This study is registered with the ISRCTN Registry, ISRCTN 25378536. FINDINGS: Between Jan 18, 2017, and April 19, 2018, 555 children were referred and 249 were eligible, agreed to participate, and were randomly assigned to either PACT-G (n=122) or treatment as usual (n=127). One child in the PACT-G group withdrew and requested their data be removed from the study, giving an ITT population of 248 children. 51 (21%) of 248 children were female, 197 (79%) were male, 149 (60%) were White, and the mean age was 4·0 years (SD 0·6). The groups were well balanced for demographic and clinical characteristics. In the PACT-G group, parents of children received a median of 10 (IQR 8-12) home sessions and LSAs received a median of 8 (IQR 5-10) education sessions over 6 months. We found no treatment effect on the ADOS-2 primary outcome compared with treatment as usual (effect size 0·04 [95% CI -0·19 to 0·26]; p=0·74), or researcher-assessed BOSCC (0·03 [-0·25 to 0·31]), language composite (-0·03 [-0·15 to 0·10]), repetitive behaviour composite (0·00 [-0·35 to 0·35]), adaptive behaviour composite (0·01 [-0·15 to 0·18]), or child wellbeing (0·09 [-0·15 to 0·34]). PACT-G treatment improved synchronous response in both parent (0·50 [0·36 to 0·65]) and LSA (0·33 [0·16 to 0·50]), mediating increased child communication with parent (0·26 [0·12 to 0·40]) and LSA (0·20 [0·06 to 0·34]). Child dyadic communication change mediated outcome symptom alteration on BOSCC at home (indirect effect -0·78 [SE 0·34; 95% CI -1·44 to -0·11]; p=0·022) although not in education (indirect effect -0·67 [SE 0·37; 95% CI -1·40 to 0·06]; p=0·073); such an effect was not seen on ADOS-2. Treatment with PACT-G also improved the parental wellbeing composite (0·44 [0·08 to 0·79]) and the child disruptive behaviour composite in home and education (0·29 [0·01 to 0·57]). Adverse events on child behaviour and wellbeing were recorded in 13 (10%) of 127 children in the treatment as usual group (of whom four [31%] were girls) and 11 (9%) of 122 in the PACT-G group (of whom three [33%] were girls). One serious adverse event on parental mental health was recorded in the PACT-G group and was possibly study related. INTERPRETATION: Although we found no effect on the primary outcome compared with treatment as usual, adaptation of the 12-month PACT intervention into briefer multicomponent delivery across home and education preserved the positive proximal outcomes, although smaller in effect size, and the original pattern of treatment mediation seen in clinic-delivered therapy, as well as improving parental wellbeing and child disruptive behaviours across home and school. Reasons for this reduced efficacy might be the reduced dose of each component, the effect of remote delivery, and the challenges of the delivery contexts. Caution is needed in assuming that changing delivery methods and context will preserve an original intervention efficacy for autistic children. FUNDING: National Institute for Health Research and Medical Research Council Efficacy and Mechanism Evaluation Award.

Autistic Self-Advocacy and the Neurodiversity Movement: Implications for Autism Early Intervention Research and Practice.

The growth of autistic self-advocacy and the neurodiversity movement has brought about new ethical, theoretical and ideological debates within autism theory, research and practice. These debates have had genuine impact within some areas of autism research but their influence is less evident within early intervention research. In this paper, we argue that all autism intervention stakeholders need to understand and actively engage with the views of autistic people and with neurodiversity as a concept and movement. In so doing, intervention researchers and practitioners are required to move away from a normative agenda and pay diligence to environmental goodness-of-fit, autistic developmental trajectories, internal drivers and experiences, and autistic prioritized intervention targets. Autism intervention researchers must respond to these debates by reframing effectiveness, developing tools to measure autistic prioritized outcomes, and forming partnerships with autistic people. There is a pressing need for increased reflection and articulation around how intervention practices align with a neurodiversity framework and greater emphasis within intervention programmes on natural developmental processes, coping strategies, autonomy, and well-being.

Language and transient emotional states affect implicit cultural bias.

Bilinguals react to cultural information in a language-dependent fashion, but it is unknown whether this is influenced by the individual's emotional state. Here, we show that induced mood states increase cultural bias-measured using the Implicit Association Test (IAT)-but this effect occurs asymmetrically across languages. In the native language, bilinguals show a strong cultural bias, which is not influenced by mood. But in the non-native language, a relatively low cultural bias significantly increases as a function of a positive or negative mood. Our findings suggest that the native language promotes an inherent cultural bias, which is impervious to fluctuations in the bilingual's mood state. In the second language, however, bilinguals are culturally impartial, unless they are in a heightened mood state.

Language and culture modulate online semantic processing.

Language has been shown to influence non-linguistic cognitive operations such as colour perception, object categorization and motion event perception. Here, we show that language also modulates higher level processing, such as semantic knowledge. Using event-related brain potentials, we show that highly fluent Welsh-English bilinguals require significantly less processing effort when reading sentences in Welsh which contain factually correct information about Wales, than when reading sentences containing the same information presented in English. Crucially, culturally irrelevant information was processed similarly in both Welsh and English. Our findings show that even in highly proficient bilinguals, language interacts with factors associated with personal identity, such as culture, to modulate online semantic processing.