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

Vanessa Abrahamson, Wenjing Zhang, Patricia Wilson, William Farr, Ian Male, Vanessa Abrahamson, Wenjing Zhang, Patricia Wilson, William Farr, Ian Male

Abstract

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.

Keywords: developmental neurology & neurodisability; health policy; primary care.

Conflict of interest statement

Competing interests: None declared.

© Author(s) (or their employer(s)) 2020. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Figures

Figure 1
Figure 1
RE-ASCeD RRR stages. RE-ASCeD, Realist Evaluation of Autism ServiCe Delivery; RRR, rapid realist review.
Figure 2
Figure 2
Developing and refining the research question.
Figure 3
Figure 3
Searching and retrieving information.
Figure 4
Figure 4
The screening process of primary search results. RAs, research assistants; RRR, rapid realist review.

References

    1. England NHS Nhs long term plan: learning disability and autism. section 3.31-3.36. England N, 2019.
    1. Baird G, Simonoff E, Pickles A, et al. . Prevalence of disorders of the autism spectrum in a population cohort of children in South Thames: the special needs and autism project (SNAP). The Lancet 2006;368:210–5. 10.1016/S0140-6736(06)69041-7
    1. Elsabbagh M, Divan G, Koh Y-J, et al. . Global prevalence of autism and other pervasive developmental disorders. Autism Res 2012;5:160–79. 10.1002/aur.239
    1. Baio J, Wiggins L, Christensen DL, et al. . Prevalence of Autism Spectrum Disorder Among Children Aged 8 Years - Autism and Developmental Disabilities Monitoring Network, 11 Sites, United States, 2014. MMWR Surveill Summ 2018;67:1–23. 10.15585/mmwr.ss6706a1
    1. Waugh I. The prevalence of autism (including Asperger’s Syndrome) in school age children in Northern Ireland 2019. Department of health ed Northern Ireland, Belfast, 2019: 1–26.
    1. American Psychiatric Association Diagnostic and statistical manual of mental disorders. Am Psychiatric Assoc 2013.
    1. Organization WH International classification of diseases XI beta draft: 6A02 autism spectrum disorder.
    1. Health and Social Care Information Centre ND Instructions and guidance notes: assuring transformation, 2018. Available: [Accessed 05 Feb 2020].
    1. Buescher AVS, Cidav Z, Knapp M, et al. . Costs of autism spectrum disorders in the United Kingdom and the United States. JAMA Pediatr 2014;168:721–8. 10.1001/jamapediatrics.2014.210
    1. Hayes SA, Watson SL. The impact of parenting stress: a meta-analysis of studies comparing the experience of parenting stress in parents of children with and without autism spectrum disorder. J Autism Dev Disord 2013;43:629–42. 10.1007/s10803-012-1604-y
    1. National initiative for autism Screening and assessment. National autism plan for children (NapC) plan for identification, assessment, diagnosis and access to early interventions for pre-school and primary school aged children with autism spectrum disorders (ASD. London, 2003: 1–134.
    1. BACCH A workforce strategy for community paediatrics. London: British association for community child health, 2019: 1–20.
    1. Care Quality Commission, Ofsted . Local area send inspections: 1 year on, 2017.
    1. Male I, Farr W, Gain A, et al. . How much does it cost to assess a child for possible autism spectrum disorder in the UK National health service: an observational study. European Academy of Childhood Disability, 2019.
    1. Rutherford M, Burns M, Gray D, et al. . Improving efficiency and quality of the children's ASD diagnostic pathway: lessons learned from practice. J Autism Dev Disord 2018;48:1579–95. 10.1007/s10803-017-3415-7
    1. Galliver M, Gowling E, Farr W, et al. . Cost of assessing a child for possible autism spectrum disorder? an observational study of current practice in child development centres in the UK. BMJ Paediatr Open 2017;1:e000052. 10.1136/bmjpo-2017-000052
    1. Brett D, Warnell F, McConachie H, et al. . Factors affecting age at ASD diagnosis in UK: no evidence that diagnosis age has decreased between 2004 and 2014. J Autism Dev Disord 2016;46:1974–84. 10.1007/s10803-016-2716-6
    1. Kentrou V, de Veld DM, Mataw KJ, et al. . Delayed autism spectrum disorder recognition in children and adolescents previously diagnosed with attention-deficit/hyperactivity disorder. Autism 2019;23:1065–72. 10.1177/1362361318785171
    1. Rutherford M, McKenzie K, Forsyth K, et al. . Why are they waiting? exploring professional perspectives and developing solutions to delayed diagnosis of autism spectrum disorder in adults and children. Res Autism Spectr Disord 2016;31:53–65. 10.1016/j.rasd.2016.06.004
    1. National Institute for Health and Care Excellence Autism spectrum disorder in under 19S: recognition, referral and diagnosis (CG128. London, UK, 2011: 1–45.
    1. Lord C, Luyster R, Guthrie W, et al. . Patterns of developmental trajectories in toddlers with autism spectrum disorder. J Consult Clin Psychol 2012;80:477–89. 10.1037/a0027214
    1. Whitehouse A, Evans K, Eapen V. The diagnostic process for children, adolescents and adults referred for assessment of autism spectrum disorder in Australia: a national guideline (draft version for community consultation. Autism CRC, 2017.
    1. Penner M, King GA, Hartman L, et al. . Community General pediatricians' perspectives on providing autism diagnoses in Ontario, Canada: a qualitative study. J Dev Behav Pediatr 2017;38:593–602. 10.1097/DBP.0000000000000483
    1. Jordan E, Farr W, Fager S, et al. . Pirate adventure autism assessment app: a new tool to aid clinical assessment of children with possible autistic spectrum disorder : Powell W, Rizzo A, Sharkey P, et al., Rehabilitation: innovation and challenges in the use of virtual reality technologies NY. US: Nova Publishers, 2017: 27–38.
    1. Male I, Reddy V. Should ADHD, ASD& related services be delivered in an integrated way? BACCH NEWS, 2018: 20–2.
    1. Crane L, Chester JW, Goddard L, et al. . Experiences of autism diagnosis: a survey of over 1000 parents in the United Kingdom. Autism 2016;20:153–62. 10.1177/1362361315573636
    1. Reed P, Osborne LA. Diagnostic practice and its impacts on parental health and child behaviour problems in autism spectrum disorders. Arch Dis Child 2012;97:927–31. 10.1136/archdischild-2012-301761
    1. Palmer E, Ketteridge C, Parr JR, et al. . Autism spectrum disorder diagnostic assessments: improvements since publication of the National autism plan for children. Arch Dis Child 2011;96:473–5. 10.1136/adc.2009.172825
    1. Saul JE, Willis CD, Bitz J, et al. . A time-responsive tool for informing policy making: rapid realist review. Implement Sci 2013;8:103. 10.1186/1748-5908-8-103
    1. Wong G. Data gathering in realist reviews: looking for needles in haystacks. Doing realist research. London: SAGE, 2018.
    1. Pawson R. Evidence-Based policy: a realist perspective. London: SAGE Publications, 2006.
    1. Willis CD, Saul JE, Bitz J, et al. . Improving organizational capacity to address health literacy in public health: a rapid realist review. Public Health 2014;128:515–24. 10.1016/j.puhe.2014.01.014
    1. Tsang JY, Blakeman T, Hegarty J, et al. . Understanding the implementation of interventions to improve the management of chronic kidney disease in primary care: a rapid realist review. Implementation Science 2015;11 10.1186/s13012-016-0413-7
    1. Weetman K, Wong G, Scott E, et al. . Improving best practise for patients receiving hospital discharge letters: a realist review protocol. BMJ Open 2017;7:e018353. 10.1136/bmjopen-2017-018353
    1. Gilmore B, McAuliffe E, Power J, et al. . Data analysis and synthesis within a realist evaluation: toward more transparent methodological approaches. Int J Qual Methods 2019;18:1609406919859754 10.1177/1609406919859754
    1. Jagosh J, Pluye P, Macaulay AC, et al. . Assessing the outcomes of participatory research: protocol for identifying, selecting, appraising and synthesizing the literature for realist review. Implement Sci 2011;6:24. 10.1186/1748-5908-6-24
    1. Wong G, Greenhalgh T, Westhorp G, et al. . RAMESES publication standards: realist syntheses. BMC Med 2013;11:21. 10.1186/1741-7015-11-21
    1. Pawson R. Digging for Nuggets: How ‘Bad’ Research Can Yield ‘Good’ Evidence. Int J Soc Res Methodol 2006;9:127–42. 10.1080/13645570600595314
    1. MacDonald M, Pauly B, Wong G, et al. . Supporting successful implementation of public health interventions: protocol for a realist synthesis. Syst Rev 2016;5:54. 10.1186/s13643-016-0229-1
    1. Jagosh J, Pluye P, Wong G, et al. . Critical reflections on realist review: insights from customizing the methodology to the needs of participatory research assessment. Res Synth Methods 2014;5:131–41. 10.1002/jrsm.1099

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