An observational study of individual child journeys through autism diagnostic pathways, and associated costs, in the UK National Health Service.

Background: Demand for diagnostic assessment in children with possible autism has recently increased significantly. Services are under pressure to deliver timely and high-quality diagnosis, following National Institute and Care Excellence multidisciplinary assessment guidelines. This UK National Health Service study aimed to answer: how many hours of health professional time are required to deliver autism diagnostic assessment, and how much does this cost?. Method: Case notes of 20 children (1-16 yrs.) from 27 NHS trusts, assessed through an autism diagnostic pathway in the previous year, were examined retrospectively. Data included: hours of professional time, diagnostic outcome. Assessment costs calculated using standardised NHS tariffs. Results: 488 children (aged 21-195 months, mean 82.9 months, SD 39.36) from 22 Child Development Services (CDS), four Child and Adolescent Mental Health Services (CAMHS) and one tertiary centre; 87% were either under 5 (36%) or 5 to 11 years (51%). Children seen by CDS were younger than CAMHS (mean (SD) 6.10 (2.72) vs. 10.39 (2.97) years, p < 0.001). Mean days to diagnosis were 375 (SD 235), with large variation (range 41-1553 days). Mean hours of professional time per child was 11.50 (SD 7.03) and varied substantially between services and individuals. Mean cost of assessment was £846.00 (SD 536.31). 339 (70.0%) children received autism diagnosis with or without comorbidity; 54 (11%) received no neurodevelopmental diagnosis; 91 (19%) received alternative neurodevelopmental diagnoses. Children with one or more coexisting conditions took longer to diagnose, and assessment was more costly, on average 117 days longer, costing £180 more than a child with no neurodevelopmental diagnosis. Age did not predict days to diagnosis or assessment costs. Conclusion: Typical assessment took 11 h of professional time and over 12-months to complete, costing GB£850 per child. Variation between centres and children reflect differences in practice and complexity of diagnostic presentation. These results give information to those delivering/planning autism assessments using multi-disciplinary team approach, in publicly funded health systems. Planning of future diagnostic services needs to consider growing demand, the need for streamlining, enabling context appropriate services, and child/family complexity.

Realist evaluation of Autism ServiCe Delivery (RE-ASCeD): which diagnostic pathways work best, for whom and in what context? Findings from a rapid realist review.

OBJECTIVES: Waiting times in the UK for an autism diagnostic assessment have increased rapidly in the last 5 years. This review explored research (including 'grey' literature) to uncover the current evidence base about autism diagnostic pathways and what works best, for whom and in what circumstances, to deliver high quality and timely diagnosis. DESIGN: We performed a Rapid Realist Review consistent with recognised standards for realist syntheses. We collected 129 grey literature and policy/guidelines and 220 articles from seven databases (January 2011-December 2019). We developed programme theories of how, why and in what contexts an intervention worked, based on cross comparison and synthesis of evidence. The focus was on identifying factors that contributed to a clearly defined intervention (the diagnostic pathway), associated with specific outcomes (high quality and timely), within specific parameters (Autism diagnostic services in Paediatric and Child & Adolescent Mental Health services in the UK). Our Expert Stakeholder Group, including representatives from local parent forums, national advocacy groups and clinicians, was integral to the process. RESULTS: Based on 45 relevant articles, we identified 7 programme theories that were integral to the process of diagnostic service delivery. Four were related to the clinical pathway: initial recognition of possible autism; referral and triaging; diagnostic model; and providing feedback to parents. Three programme theories were pertinent to all stages of the referral and diagnostic process: working in partnership with families; interagency working; and training, service evaluation and development. CONCLUSIONS: This theory informed review of childhood autism diagnostic pathways identified important aspects that may contribute to efficient, high quality and family-friendly service delivery. The programme theories will be further tested through a national survey of current practice and in-depth longitudinal case studies of exemplar services. TRIAL REGISTRATION NUMBER: NCT04422483.

Feasibility of a randomised controlled trial to evaluate home-based virtual reality therapy in children with cerebral palsy.

PURPOSE: Evidence is increasing for effective virtual reality therapy for motor rehabilitation for children with Cerebral Palsy. We assessed the feasibility of a virtual reality therapy mode of intervention, appropriateness of measures, and potential cost-effectiveness. METHODS: A 12-week, 2-group, parallel-feasibility trial (ISRCT 17624388) using Nintendo Wii FitTM at home. Children aged 5-16, with ambulatory Cerebral Palsy, who were able to follow simple instructions were randomised to two groups; one supported by physiotherapists (individualised activity programme), the other unsupported with children having free choice (control). Children were assessed in clinic at baseline, week 6, and week 12 by blinded assessors. Feasibility of the intervention was assessed via recruitment, adherence, and usefulness of measurement tools. RESULTS: Forty-four children were eligible (out of 48 approached): 31 consented, 30 were randomised, 21 completed the study; 10 in the supported group and 11 in the unsupported group. Nine children discontinued from tiredness, after-school activities, homework, surgery, technical difficulties or negative system feedback. The supported group completed 19 of 36 (IQR 5-35) possible sessions; the unsupported group 24 of 36 sessions (IQR 8-36). Gross Motor Function Measure scores varied by Cerebral Palsy severity after the intervention. There were no adverse events. CONCLUSION: Virtual reality therapy offers potential as a therapeutic adjunct for children with Cerebral Palsy, warranting substantive confirmatory study. Gross Motor Function Measure, with modifications to improve sensitivity, appeared appropriate as a primary measure, with Timed up and Go test secondary. The intervention was inexpensive costing £20 per child. An explanatory trial to evaluate the clinical/cost-effectiveness of commercial system virtual reality therapy is feasible with minor methodological adaptation. Implications for rehabilitation Home-based interactive computer gaming was feasible, safe and cost effective as a therapy adjunct. Discontinue if additional pressures are present: imminent surgery, family resilience to technical difficulties, negative system feedback, after-school activities. Change in Gross Motor Function Measurement scores varied by severity of Cerebral Palsy.

Realist Evaluation of Autism ServiCe Delivery (RE-ASCeD): which diagnostic pathways work best, for whom and in what context? Protocol for a rapid realist review.

INTRODUCTION: The National Health Service (NHS) Long-Term Plan (2019) acknowledges that children and young people with suspected autism wait too long for diagnostic assessment and sets out to reduce waiting times. However, diagnostic pathways vary with limited evidence on what model works best, for whom and in what circumstances. The National Autism Plan for Children (2003) recommended that assessment should be completed within 13 weeks but referral to diagnosis can take as long as 799 days.This Rapid Realist Review (RRR) is the first work package in a national programme of research: a Realist Evaluation of Autism ServiCe Delivery (RE-ASCeD). We explore how particular approaches may deliver high-quality and timely autism diagnostic services for children with possible autism; high quality is defined as compliant with National Institute for Heath and Care Excellence (2011) guidelines, and timely as a pathway lasting no more than one calendar year, based on previous work. METHODS AND ANALYSIS: RRR is a well-established approach to synthesising evidence within a compressed timeframe to identify models of service delivery leading to desired outcomes. RRR works backwards from intended outcomes, identified by NICE guidelines and the NHS England Long-Term Plan. The focus is a clearly defined intervention (the diagnostic pathway), associated with specific outcomes (high quality and timely), within a particular set of parameters (Autism and Child & Adolescent Mental Health services in the UK). Our Expert Stakeholder Group consists of policymakers, content experts and knowledge users with a wide range of experience to supplement, tailor and expedite the process. The RRR is consistent with Realist And Meta-narrative Evidence Syntheses: Evolving Standards (RAMESES) and includes identifying the research question, searching for information, quality appraisal, data extraction, synthesising the evidence, validation of findings with experts and dissemination. ETHICS AND DISSEMINATION: Ethical approval not required. Findings will inform the wider RE-ASCeD evaluation and be reported to NHS England. TRIAL REGISTRATION NUMBER: NCT04422483. This protocol relates to Pre-results.

Cost of assessing a child for possible autism spectrum disorder? An observational study of current practice in child development centres in the UK.

OBJECTIVE: UK guidelines recommend that diagnosis of autism in children requires assessment by a multidisciplinary team. With growing numbers of referrals for assessment, diagnostic services have been under increasing pressure to meet the level of need. This study aimed to explore the number of hours of professional time required to complete such an assessment based on current practice in secondary care child development centres across the UK, and from this we calculate the cost of assessment. DESIGN: An online questionnaire, using SurveyMonkey.com, was sent to 20 child development centres asking them to retrospectively record team members involved at each stage of assessment and time taken, including report writing and administration for a typical assessment. Costs were estimated based on the hourly rate for each team member, including salary, on-costs and trust overheads. RESULTS: 12 questionnaires (60%) were returned. 10 centres adopted a two-stage approach to assessment with an initial 'screening' clinic determining whether the child needed to proceed to full multidisciplinary assessment. Median professional time involved was 13 hours (IQR 9.6-15.5 hours). This resulted in a median cost of £809 ($1213, based on conversion rate £1 equal to US$1.5 (November 2015)), (IQR £684-£925) ($1026-$1388)). IMPLICATIONS: This study confirms that multidisciplinary diagnostic assessment of a child with possible autism requires significant professional time, with staff costs of approximately £800 ($1200) per child. This does not include costs of intervention, parent psychological education, investigation and assessment and management of comorbidities. If growing waiting times for diagnostic assessment are to be avoided, funding for diagnostic services needs to reflect the human resources required and the resulting costs of that assessment.

Congenital lower brachial plexus palsy due to cervical ribs.

Congenital brachial plexus palsy (CBPP) usually occurs secondarily to intrapartum trauma, but this is not always the case. Cervical ribs have previously been reported to increase the risk of CBPP in association with birth trauma. We report the cases of two children (one female, one male) with congenital lower brachial plexus palsy in whom the presence of non-ossified cervical ribs was the only identified risk factor. In the female child magnetic resonance imaging (MRI) of the brain, spinal cord, and brachial plexus revealed no abnormality except for the presence of bilateral cervical ribs at the level of the seventh cervical (C7) vertebra. Chest radiography was normal, which suggested that the cervical ribs identified on the MRI were fibrous bands or cartilaginous ribs rather than ossified ribs. In the male child, MRI of the spine and brachial plexus was normal but he was noted to have bilateral cervical ribs at C7. These were not identifiable on chest radiography and, therefore, are likely to reflect fibrous bands or cartilaginous ribs.