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Background: Autistic people commonly have physical and mental health conditions. They also frequently experience barriers to accessing healthcare, contributing to problems identifying and treating health conditions. These factors may lead to increased and earlier morbidity and lower average life expectancy for autistic people. Health checks specifically designed for autistic people, incorporating adjustments to healthcare, may help to overcome these barriers and reduce health inequalities. This trial aims to investigate the clinical and cost-effectiveness of a primary care health check for autistic adults and explore factors related to implementation such as acceptability and feasibility of delivery. The trial is co-designed and delivered by health professionals, autistic people, carers and supporters, and researchers., Methods: This is a clinical and cost-effectiveness, cluster randomised controlled trial of a primary care health check for autistic adults. Primary care practices will be randomised into one of two groups (intervention or control). Two hundred autistic adults (aged 18 years and over) who provide baseline data will be recruited via participating practices. Data will be collected through quantitative and qualitative methods. The primary outcome will be the incidence of new health needs/conditions detected and met at 9 months (data gathered from participant's GP records). Secondary outcomes will include the following: cost-effectiveness, measured as incremental cost per quality-adjusted life year gained over 9 months; the extent of health monitoring and health promotion needs met at 9 months; the incidence of social care needs identified at 9 months; changes in participant or carer general health; changes in quality of life; primary and secondary health and social care resource usage and costs. A qualitative study will explore views about the acceptability of the health check, its utility and future use., Discussion: This study will examine the effectiveness and cost-effectiveness of a primary care health check for autistic adults in identifying new health conditions and needs. If the intervention is effective, it would provide strong evidence for implementation into routine healthcare, therefore enabling earlier health condition diagnosis and opportunities for treatment, reducing the health inequalities experienced by autistic people., Trial Registration: ISRCTN, retrospectively registered on 20 July 2023. https://www.isrctn.com/ISRCTN30156776 (ISRCTN registration number: 30156776)., Competing Interests: Declarations. Ethics approval and consent to participate: This study received ethical approval from the North Wales Research Ethics Committee (Wales REC 5) and the NHS Health Research Authority (ref: 21/WA/0196; IRAS Project ID: 272808). Informed consent will be obtained from all study participants. Important protocol modifications will be reported in writing to relevant parties. A list of all protocol modifications will be provided during study reporting. Consent for publication: Not applicable. Competing interests: The authors declare that they have no competing interests. Primary care practices are receiving funding for research activities at sites. To the best of our knowledge, primary care sites have no competing or financial interests., (© 2024. The Author(s).)

Objectives: In England, the delivery of health, education and social care services changed substantially during COVID-19. Some services closed, some had reduced capacity and there was a shift to the use of telehealth. This study aimed to understand how families of children and young people with neurodisability experienced these service changes, what did or did not work well for them and what impact the service changes had on them., Design: Qualitative study with parent carers of children (aged 0-19 years) with neurodisability accessing children's health, social care and education-based services during the COVID-19 pandemic., Setting: Participants were recruited from five local authority areas in England and interviewed via telephone and/or video call., Participants: 48 parent carers (45 mothers, three fathers) were interviewed and nine young people (aged 8-16 years). Across the parent carers there were 55 children with neurodisability (43 males, 12 females), ranging from 3 to 19 years. Children had a range of diagnoses, including autism, attention deficit hyperactivity disorder, cerebral palsy, genetic conditions and epilepsy. Nine young people (aged 8-16 years; eight males, one female) were interviewed; two individually, three in a focus group and four with their parent carer., Results: Four themes were identified: (1) communication of service changes, (2) access to services during the pandemic, (3) impacts of service changes and (4) learning for future emergencies and resetting services. Communication of service changes was reported as poor and confusing. Access to services during the pandemic varied. Medical services continued with least disruption; therapeutic, education-based and social care services were severely disrupted. Service changes had a detrimental impact on families coping with high levels of medical care and physical and behavioural support. Young people experienced negative impacts of service change on their physical, mental and behavioural health., Conclusions: Services for children with neurodisability require a person-led, family centred approach with strong multidisciplinary team working. Findings indicated the need for improved communication within and between services, and between services and families and young people. Planning for future emergencies needs to factor in the specific health and care needs of children with neurodisability and maintain access to services, in particular, those accessed through schools., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2024. Re-use permitted under CC BY. Published by BMJ.)

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., (© 2023. The Author(s).)

Aim: To seek consensus on recommendations for the delivery of services to disabled children in England during future emergencies., Method: Candidate recommendations were drafted based on our related mapping review and qualitative research related to experiences during the COVID-19 pandemic. Iterative workshops with professionals and parent carers helped to refine the recommendations. A Delphi survey, rating the importance of each recommendation, was conducted with (1) parent carers of disabled children, (2) disabled young people aged 8 to 19 years, and (3) health, education, and social care professionals. A consensus meeting was convened online to discuss the findings and ratify the recommendations., Results: Twenty-eight recommendations were included in the Delphi survey. There were 141 participants in round 1 and 91 in round 2. Seven recommendations reached the agreed consensus criteria for being critical across all stakeholder groups, while 21 recommendations did not reach consensus across all groups. Fourteen participants ratified 23 recommendations, which when aggregated and refined further produced our final 19 recommendations., Interpretation: Reductions in services for disabled children and their families during the COVID-19 pandemic had serious and lasting consequences. This study enabled parent carers, disabled young people, and health, education, and social care professionals to agree recommendations on services for disabled children during future emergencies., (© 2024 The Author(s). Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.)

Objectives: To understand how health, education and social care services for disabled children changed during the COVID-19 pandemic, what did or did not work well and what the impacts of service changes were on both professionals and families., Design: Qualitative study using semistructured interviews., Setting: Telephone and video call interviews and focus groups with professionals working in one of five local authority areas in England., Participants: 78 health, education and social care professionals working with children in one of five local authority areas in England., Results: There was a significant disruption to services and reduced contact with families during the early stages of the pandemic; nevertheless, professionals were able to reflect on innovative ways they interacted with and sought to support and maintain health, education and social care provision to disabled children and their families. As waitlists have substantially increased, this and the longevity of the pandemic were perceived to have had negative consequences for staff health and well-being, the health and psychosocial outcomes of children and young people, and their parent carers., Conclusions: Key learning from this study for service recovery and planning for future emergencies is the need to be able to identify disabled children, classify their level of need and risk, assess the impact of loss of services and maintain clear communication across services to meet the needs of disabled children. Finally, services need to work collaboratively with families to develop child-centred care to strengthen resilience during service disruption., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2024. Re-use permitted under CC BY. Published by BMJ.)

Objectives: Healthcare professionals (HCPs) need to identify potentially serious musculoskeletal (MSK) presentations in children and refer them to specialists appropriately. Our aim was to develop 'pGALSplus' (paediatric gait, arms, legs and spine plus) to support clinical assessment, aid decision-making and assess feasibility and acceptability in exemplar MSK pathologies., Methods: We used a three-phase mixed methods approach: phase 1, preliminary stakeholder engagement and scoping review to propose pGALSplus; phase 2, iterative development of pGALSplus involving an expert working group; and phase 3, testing the feasibility of pGALSplus in exemplar MSK conditions [JIA, mucopolysaccharidoses (MPS), muscular dystrophy (MD), developmental coordination disorder (DCD) and healthy controls (HCs)]. The final pGALSplus was derived from analysis of phase 3 data and feedback from HCPs, families and expert consensus input from an international e-survey ( n  = 22) and virtual event ( n  = 13)., Results: Feasibility was tested in 45 children (JIA, n  = 10; MPS, n  = 6; MD, n  = 9; DCD, n  = 10; HCs, n  = 10). Overall the assessment was achievable in the target age range (2-10 years) and quick to complete [median 12 min (range 8-20)], with high acceptability from families. Expert feedback deemed pGALSplus to be very useful and of particular use to non-specialists in MSK paediatrics. The final pGALSplus comprises 26 clinical observations/skills with a colour-coding approach to aid decision-making and identification of more serious MSK presentations and additional resources to support its use in clinical practice., Conclusions: pGALSplus is a novel evidence- and consensus-based assessment building on pGALS, with high acceptability and feasibility. As community-based MSK assessment in children becomes more established, we propose that pGALSplus will facilitate and inform decision-making to promote access to specialist care., (© The Author(s) 2024. Published by Oxford University Press on behalf of the British Society for Rheumatology.)

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., Competing Interests: The authors have declared that no competing interests exist., (Copyright: © 2024 Hamilton et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.)

Background: Anxiety related to uncertainty is common in autism. Coping with Uncertainty in Everyday Situations (CUES©) is a parent-mediated group intervention aiming to increase autistic children's tolerance to uncertain situations. A pilot study was conducted to test its feasibility and acceptability., Methods: Parents of 50 autistic children were randomised to receive CUES© or enhanced services as usual., Results: All children met the clinical threshold for at least one anxiety disorder. Of the 26 participants randomised to CUES©, 72% attended 4-8 sessions. Parents and therapists reported they found CUES© useful and acceptable., Conclusions: Families were willing to be recruited and randomised, the format/content was feasible to deliver, and the outcome measures were acceptable. CUES© should be evaluated in a clinical and cost effectiveness randomised controlled trial., (© 2022. The Author(s).)

Adaptive functioning of autistic children is traditionally measured through informant-report, often from parents. Behaviour varies across settings though, and context-specific reports should be considered. Limited and inconsistent results show low parent-education professional concordance, but no research has yet explored item level response variation. We investigated Vineland Adaptive Behaviour Scales-II concordance using 233 lower ability autistic children from the PACT-G sample. Domain and item level agreement was low, but better on objectively measured behaviours. Higher child nonverbal ability improved concordance. Where disagreements occurred, education professionals identified emergent skills more and parents were more likely to rate present/absent. Parents and education professionals view the adaptive abilities of autistic children differently and both should be considered when developing personalised interventions and support., (© 2022. The Author(s).)

Aim: To identify the research on childhood disability service adaptations and their impact on children and young people with long-term disability during the COVID-19 pandemic., Method: A mapping review was undertaken. We searched the World Health Organization Global COVID-19 database using the search terms 'children', 'chronic/disabling conditions', and 'services/therapies'. Eligible papers reported service changes for children (0-19 years) with long-term disability in any geographical or clinical setting between 1st January 2020 and 26th January 2022. Papers were charted across the effective practice and organization of care taxonomy of health system interventions and were narratively synthesized; an interactive map was produced., Results: Reduction of face-to-face care and usual provision had a huge impact on children and families. Adoption of telehealth provided continuity for the care and management of some conditions. There was limited evidence of changes to mental health services, transitions of care, social care, or child-reported satisfaction or acceptability of service changes., Interpretation: The long-term impacts of service change during the pandemic need full evaluation. However, widespread disruption seems to have had a profound impact on child and carer health and well-being. Service recovery needs to be specific to the individual needs of children with a disability and their families. This should be done through coproduction to ensure that service changes meet needs and are accessible and equitable., (© 2023 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.)

Lay Abstract: Autistic people are on average more likely to experience poor health than people who are not autistic. Health checks have been shown to improve access to effective healthcare. This study investigated people's views about a primary care health check for autistic adults. We held discussion groups and interviewed autistic adults, adults with intellectual disabilities, supporters and health professionals. People wanted the health check to look at a person's physical and mental health, and how they were doing socially. They thought people should be able to share information about their needs and the reasonable adjustments they would like before the health check. They wanted healthcare services to change the way they communicate with autistic people, such as being able to book appointments online rather than by telephone. They wanted a choice in how the health check was completed, with video call or email offered as well as face-to-face appointments. People thought further training of primary care staff on autism was needed, to increase awareness of the diversity of experiences of autistic people and ways in which difficulties, such as pain, may present differently to non-autistic people. Clinicians raised questions about whether mental health and social care services could meet the additional needs that might be identified through the health check. We used this information to design an NHS primary care health check for autistic people in collaboration with autistic people, supporters and health professionals.

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.

Background: Compared with the general population, autistic adults experience higher rates of physical and mental health conditions, premature morbidity and mortality, and barriers to health care. A health check for autistic people may improve their health outcomes., Aim: To establish the views of autistic people towards a primary care health check for autistic people., Design & Setting: Cross-sectional questionnaire study in England and Wales., Method: A questionnaire was sent to autistic adults with physical health conditions in England and Wales. A total of 458 people (441 autistic adults and 17 proxy responders) completed the questionnaire., Results: Most responders (73.4%, n = 336) thought a health check is needed for all autistic people. Around half of the participants thought a health check should be offered from childhood and the health check appointment should last between 15 and 30 minutes. Autistic people were positive about providing primary care staff with contextual information regarding their health and the reasonable adjustments they would like before their health check appointment. Training about autism and the health check was considered important, alongside adequate time for discussions in the health check appointment (all by over 70% of responders). The clinician's autism knowledge, seeing a familiar clinician, environmental adaptations, appropriate information, and accessible appointments were considered particularly important in making a health check accessible., Conclusion: Autistic people and relatives were supportive of a primary care health check for autistic people. Information gathered was used to support the design of a primary care health check for autistic adults., (Copyright © 2022, The Authors.)

Lay Abstract: Living with undiagnosed autism can be distressing and may affect mental health. A diagnosis of autism can help self-awareness and self-understanding. However, it can be difficult for adults to access an autism assessment. Clinicians also sometimes find it hard to identify autism in adults. This may mean an autism diagnosis is delayed or missed. In this study, we asked autistic adults, relatives and clinicians how to improve this. The study was in two stages. In the first stage (stage 1), 343 autistic adults and 45 relatives completed a survey. In the survey, we asked questions about people's experiences of UK autism assessment services for adults. Thirty-five clinicians completed a similar survey. Clinicians reported that some autism assessment teams lacked key professionals, for example, psychologists and occupational therapists. We used the information from the three separate surveys to create 13 statements describing best autism assessment services for adults. In stage 2, we asked clinicians for their views on the 13 statements. Clinicians agreed with 11 of the statements. Some autistic adults, relatives and clinicians were positive about autism assessment services, and many also described areas that could be improved. The study findings can be used to improve UK adult autism assessment services and may be helpful for service developments worldwide.

Distress caused by sensory processing differences for autistic individuals may be reduced by repetitive behaviours (RRB), including repetitive motor (RMB) and insistence on sameness (ISB) behaviours. Intolerance of uncertainty (IU) and anxiety mediate the relationship between sensory processing and RRB in autistic children. We replicated this model in autistic adults, extending it to include alexithymia. Serial mediation, using data from 426 autistic adults, identified significant direct effects from sensory processing to RMB and ISB, and indirect effects through alexithymia-IU-anxiety for RMB, and IU alone, and alexithymia-IU for ISB. Different mechanisms may underpin RMB and ISB. Alexithymia alongside, IU and anxiety, should be considered when understanding the relationship between sensory processing and RRB, and when offering interventions to support autistic people., (© 2021. The Author(s).)

Lay Abstract: Anxiety is common in autistic children. Research shows that this may be related to intolerance of uncertainty, which is a tendency to react negatively to uncertain situations. Understanding when, why and how autistic children respond to uncertainty is important in the development of anxiety programmes. We asked 53 (including 3 dyads) parents of autistic children about the types of uncertain situations that cause difficulties for their child and how uncertainty impacts on daily life for them and their families. We found that uncertain situations made autistic children and their families feel sad, worried, frustrated and angry through the themes: child's reactions to uncertainty, trying to reduce uncertainty, the impact of difficulties with uncertainty, the impact of uncertainty on parenting and the impact on parents. There are lots of situations that are anxiety provoking for autistic children because of uncertainty, such as school. Programmes to reduce anxiety and increase autistic children's ability to cope with everyday uncertain situations could improve quality of life for autistic children and their families.

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., Competing Interests: The authors have declared that no competing interests exist.

Aim: To investigate whether children with perinatal brain injury have impairments in specific components of visual attention, and whether early dietary supplementation can reduce any deficits., Method: Children participating in the Dolphin neonatal trial of dietary supplementation were tested at age 6 months with the Infant Fixation Shift Attention Test, and at 4 to 5 years with four subtests of the Early Childhood Attention Battery (ECAB) assessing different components of attention (selective, sustained, and executive function), and the Fluid Crystallized Intelligence Index of the Kaufman Assessment Battery for Children, Second Edition (KABC-II). From 59 children originally assigned to trial groups, 33 were available for testing at 4 to 5 years (18 treatment group of whom seven, six, and five showed mild, moderate, or severe neonatal brain injury; 15 controls with one, seven, and seven in the neonatal brain injury categories respectively). Given the imbalance in numbers with mild brain injury, analysis of trial group differences is restricted to moderate and severe brain injury severities (n=25)., Results: Children with perinatal brain injury showed poorer attention across all components relative to age norms (mean standard scores 75-87; p<0.001 for three of the four subtests), with the greatest impairment in sustained attention. These impairments remained when compared with cognitive age assessed using the Fluid Crystallized Intelligence Index. Impairment was reduced in the treatment compared to the control group (p=0.04 for flanker test, p=0.002 for counterpointing, and p=0.027 for the overall ECAB score)., Interpretation: Perinatal brain injury is associated with later impaired attention, beyond that predicted from any general cognitive disability. Impairment varies across attention components, being most severe for sustained attention. The effects on flanker and counterpointing suggest that dietary supplementation from 0 to 2 years of age may reduce attention problems. Measuring the different components of attention is important when considering assessment and interventions for children with perinatal brain injury., (© 2021 Mac Keith Press.)