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

Vanessa Abrahamson, Wenjing Zhang, Patricia M Wilson, William Farr, Venkat Reddy, Jeremy Parr, Anna Peckham, Ian Male, Vanessa Abrahamson, Wenjing Zhang, Patricia M Wilson, William Farr, Venkat Reddy, Jeremy Parr, Anna Peckham, Ian Male

Abstract

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.

Keywords: developmental neurology & neurodisability; paediatric neurology; primary care; quality in health care.

Conflict of interest statement

Competing interests: None declared.

© Author(s) (or their employer(s)) 2021. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Figures

Figure 1
Figure 1
Search and review flow diagram.
Figure 2
Figure 2
Programme theories (PTs) for the autism diagnostic pathway.

References

    1. Rodgers H, McCluney J. The prevalence of autism (including Asperger syndrome) in school age children in Northern Ireland 2021. Belfast, Northern Ireland: Information & Analysis Directorate, Department of Health, 2021.
    1. Roman-Urrestarazu A, van Kessel R, Allison C, et al. . Association of race/ethnicity and social disadvantage with autism prevalence in 7 million school children in England. JAMA Pediatr 2021;175:e210054-e. 10.1001/jamapediatrics.2021.0054
    1. Maenner MJ, Shaw KA, Baio J, et al. . Prevalence of autism spectrum disorder among children aged 8 years — autism and developmental disabilities monitoring network, 11 sites, United States, 2016. MMWR Surveill. Summ. 2020;69:1–12. 10.15585/mmwr.ss6904a1
    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. Wigham S, Male I, Farr W. UK childhood autism diagnostic services survey 2020: challenges and innovations. Archives of Disease in Childhood. In Press 2021.
    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. NHS England . NHS long term plan: learning disability and autism. section 3.31-3.36, 2019. Available:
    1. American Psychiatric Association . Diagnostic and statistical manual of mental disorders. Fifth ed. Arlington, VA: American Psychiatric Association, 2013.
    1. Simonoff E, Pickles A, Charman T, et al. . Psychiatric disorders in children with autism spectrum disorders: prevalence, comorbidity, and associated factors in a population-derived sample. J Am Acad Child Adolesc Psychiatry 2008;47:921–9. 10.1097/CHI.0b013e318179964f
    1. Joshi G, Petty C, Wozniak J, et al. . The heavy burden of psychiatric comorbidity in youth with autism spectrum disorders: a large comparative study of a psychiatrically referred population. J Autism Dev Disord 2010;40:1361–70. 10.1007/s10803-010-0996-9
    1. Wistow R, Barnes D. A profile of child and adolescent mental health services in England 2007/8, 2009. Available:
    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. 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. Male I, Farr W, Gain A. 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. Vanhaecht K, Sermeus W, Van Zelm R. Care pathways are defined as complex interventions. BMC Medicine 2010;8:31.
    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. Male I, Reddy V. Should ADHD, ASD & related services be delivered in an integrated way? BACCHNEWS, 2018.
    1. Male I, Farr W, Reddy V. Autism or related conditions be delivered in an integrated neurodevelopmental pathway? J Integr HealthCare 2020;2:e000037.
    1. Abrahamson V, Zhang W, Wilson P, et al. . 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. BMJ Open 2020;10:e037846. 10.1136/bmjopen-2020-037846
    1. RCPCH . Invited reviews the first four years, 2017. Available:
    1. BACCH . A workforce strategy for community paediatrics. London: British association for community child health, 2019.
    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. NIHR Centre for Engagement and Dissemination . Uk standards for public involvement, 2021. Available:
    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. Williams AC. Autoextraction of twelve permanent teeth in a child with autistic spectrum disorder. Int J Paediatr Dent 2016;26:157–9. 10.1111/ipd.12161
    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. 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. Pearson M, Brand SL, Quinn C, et al. . Using realist review to inform intervention development: methodological illustration and conceptual platform for collaborative care in offender mental health. Implement Sci 2015;10:134. 10.1186/s13012-015-0321-2
    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. Abbott M, Bernard P, Forge J. Communicating a diagnosis of Autism Spectrum Disorder - a qualitative study of parents' experiences. Clin Child Psychol Psychiatry 2013;18:370–82. 10.1177/1359104512455813
    1. O’Reilly M, Nina Lester J, Muskett T, et al. . How parents build a case for autism spectrum disorder during initial assessments: ‘We’re fighting a losing battle’. Discourse Stud 2017;19:69–83. 10.1177/1461445616683590
    1. Potter CA. “I received a leaflet and that is all”: Father experiences of a diagnosis of autism. Br J Learn Disabil 2017;45:95–105. 10.1111/bld.12179
    1. Dowden A. Improving the diagnosis of autism spectrum disorder. Prescriber, 2018: 28–30.
    1. Hurt L, Langley K, North K, et al. . Understanding and improving the care pathway for children with autism. Int J Health Care Qual Assur 2019;32:208–23. 10.1108/IJHCQA-08-2017-0153
    1. The Scottish Government . Scottish strategy mapping report, 2014. Available:
    1. Unigwe S, Buckley C, Crane L, et al. . GPs’ confidence in caring for their patients on the autism spectrum: an online self-report study. Br J Gen Pract 2017;67:e445–52. 10.3399/bjgp17X690449
    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. McKenzie K, Forsyth K, O'Hare A, et al. . Factors influencing waiting times for diagnosis of autism spectrum disorder in children and adults. Res Dev Disabil 2015;45-46:300–6. 10.1016/j.ridd.2015.07.033
    1. Tollerfield I, Pearce H. Use of the thinking patterns in autism profiling model within a diagnostic assessment service for autism. Good Autism Practice 2020;21:99–117.
    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. Autistica . Embracing complexity in diagnosis: multi diagnostic pathways for NDCs, 2019. Available:
    1. GOSH Foundation Trust . Neurodevelopmental assessment clinic (including the autism assessment service. Contract No.: Ref: 2018F0719. London, 2018.
    1. Cumbria Northumberland Tyne and Wear NHS Foundation Trust . Complex neurodevelopmental disorders service (CNDS) for children and young people, 2018. Available:
    1. Carpenter P. Diagnosis and assessment in autism spectrum disorders. Adv Ment Health Intellect Disabil 2012;6:121–9. 10.1108/20441281211227184
    1. Male I, Farr W, Gowling E. How much does it cost the NHS to assess a child for possible autism: a prospective study – Paul Polani Award Lecture. British Academy of Childhood Disability Annual Conference, 2018.
    1. Karim K, Cook L, O'Reilly M. Diagnosing autistic spectrum disorder in the age of austerity. Child Care Health Dev 2014;40:115–23. 10.1111/j.1365-2214.2012.01410.x
    1. Gray L, Gibbs J, Jolleff N, et al. . Variable implementation of good practice recommendations for the assessment and management of UK children with neurodisability. Child Care Health Dev 2015;41:938–46. 10.1111/cch.12272
    1. Halpin J. What do nurses think they are doing in pre-school autism assessment? Br J Nurs 2016;25:319–23. 10.12968/bjon.2016.25.6.319
    1. Rogers CL, Goddard L, Hill EL, et al. . Experiences of diagnosing autism spectrum disorder: a survey of professionals in the United Kingdom. Autism 2016;20:820–31. 10.1177/1362361315611109
    1. Juárez AP, Weitlauf AS, Nicholson A, et al. . Early identification of ASD through telemedicine: potential value for underserved populations. J Autism Dev Disord 2018;48:2601–10. 10.1007/s10803-018-3524-y
    1. Tryfona C, Oatley G, Calderon A, Antona M, Stephanidis C, eds. M-Health solutions to support the National health service in the diagnosis and monitoring of autism spectrum disorders in young children. Universal Access in Human-Computer Interaction: Users and Context Diversity, Pt Iii. Lecture Notes in Computer Science, 2016: 249–56.
    1. Jordan E, Farr W, Fager S. Pirate adventure autism assessment app: a new tool to aid clinical assessment of children with possible autistic spectrum disorder. In: Powell W, Rizzo A, Sharkey P, eds. Rehabilitation: innovation and challenges in the use of virtual reality technologies. NY, US: Nova Publishers, 2017: 27–38.
    1. QbTech . ADHD tests, 2020. Available:
    1. Lord C. Assessments in this time of social distancing, 2020. Available:
    1. Hennel S, Coates C, Symeonides C, et al. . Diagnosing autism: Contemporaneous surveys of parent needs and paediatric practice. J Paediatr Child Health 2016;52:506–11. 10.1111/jpc.13157
    1. Healthcare Improvement Scotland . Sign 145: assessment, diagnosis and interventions for autism spectrum disorders [1-83], 2016. Available:
    1. Crane L, Batty R, Adeyinka H, et al. . Autism diagnosis in the United Kingdom: perspectives of autistic adults, parents and professionals. J Autism Dev Disord 2018;48:3761–72. 10.1007/s10803-018-3639-1
    1. Gregory S, Arnold T, Sharman C. The development of a child and adolescent mental health service for children with disabilities: rationale for the approach, method and techniques. Good Autism Practice 2013;14:74–9.
    1. National Institute for Health and Care Excellence . CG128 Surveillance report - recommendation for guidance executive 2014a [1-98]. Available:
    1. Gregory S, Arnold T, Sharman C. The development of a child and adolescent mental health service specifically for children with disabilities: reflections on the first four years. Good Autism Practice 2013;14:68–73.
    1. Calzada LR, Pistrang N, Mandy WP, et al. . and Asperger’s disorder: Utility and meaning for families. Journal of Autism and Developmental Disorders 2012;42:230–43.
    1. Autism ACHIEVE Alliance . ASD: Waiting for Assessment - Executive Summary, 2014. Available:
    1. The Scottish Government . The Scottish Strategy for Autism - Engagement Analysis 2018 Edinburgh, 2018. Available:
    1. Health Education England . Advanced clinical practice: capabilities framework when working with people who have a learning disability and/or autism, 2020. Available:
    1. Allard A, Fellowes A, Gardiner A, et al. . It takes leaders to break down siloes. integrating services for disabled children, 2019. Available:
    1. National Institute for Health and Care Excellence . Qs 51 autism quality standard 2014b. Available:
    1. Penner M, Anagnostou E, Andoni LY, et al. . Systematic review of clinical guidance documents for autism spectrum disorder diagnostic assessment in select regions. Autism 2018;22:10.1177/1362361316685879:517–27. 10.1177/1362361316685879
    1. Whitehouse A, Evans K, Eapen V, et al. . A national guideline for the assessment and diagnosis of autism spectrum disorders in Australia, 2018. Available:
    1. van der Meer L, Evans K. The autism diagnostic process in New Zealand: final report. Brisbane Autism CRC, 2021.
    1. Williams AN, Mold B, Kilbey L, et al. . Forty years of referrals and outcomes to a UK child development centre (CDC): has demand plateaued? Child Care Health Dev 2018;44:10.1111/cch.12552:364–9. 10.1111/cch.12552
    1. Gordon-Lipkin E, Foster J, Peacock G. Whittling down the wait time: exploring models to minimize the delay from initial concern to diagnosis and treatment of autism spectrum disorder. Pediatr Clin North Am 2016;63:851–9. 10.1016/j.pcl.2016.06.007
    1. Swanson AR, Warren ZE, Stone WL, et al. . The diagnosis of autism in community pediatric settings: does advanced training facilitate practice change? Autism 2014;18:555–61. 10.1177/1362361313481507
    1. Sohl K, Mazurek MO, Brown R. Echo autism: using technology and mentorship to bridge gaps, increase access to care, and bring best practice autism care to primary care. Clin Pediatr 2017;56:509–11. 10.1177/0009922817691825
    1. NHSE and NHSI autism program strategy group . Autism diagnosis and support deep dives. NHS England and NHS Improvement, 2020.
    1. Salomone E, Pacione L, Shire S, et al. . Development of the who caregiver skills training program for developmental disorders or delays. Front Psychiatry 2019;10:769. 10.3389/fpsyt.2019.00769
    1. Reddy V, Brumpton L. Digital-driven service improvement during the COVID-19 pandemic. Paediatrics and Child Health, 2021: 1–3.
    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. Ford T, Kenchington R, Norman S, et al. . The agreement between the referrer, practitioner and research diagnosis of autistic spectrum conditions among children attending child and adolescent mental health services. Eur Child Adolesc Psychiatry 2019;28:1253–64. 10.1007/s00787-019-01290-z
    1. McKenzie K, Forsyth K, O'Hare A, et al. . The relationship between waiting times and 'adherence' to the Scottish Intercollegiate Guidelines Network 98 guideline in autism spectrum disorder diagnostic services in Scotland. Autism 2016;20:395–401. 10.1177/1362361315586136
    1. Ahlers K, Gabrielsen TP, Ellzey A, et al. . A pilot project using pediatricians as initial diagnosticians in multidisciplinary autism evaluations for young children. J Dev Behav Pediatr 2019;40:1–11. 10.1097/DBP.0000000000000621
    1. RASDN . Six Steps of Autism Care - for Children and Young People in Northern Ireland, 2011. Available:
    1. Reed P, Picton L, Grainger N, et al. . Impact of diagnostic practices on the self-reported health of mothers of recently diagnosed children with ASD. Int J Environ Res Public Health 2016;13:07. 10.3390/ijerph13090888
    1. Hayes J, Ford T, Rafeeque H, et al. . Clinical practice guidelines for diagnosis of autism spectrum disorder in adults and children in the UK: a narrative review. BMC Psychiatry 2018;18:1–25. 10.1186/s12888-018-1800-1
    1. NHS Education for Scotland . The NHS Education for Scotland Autism Training Framework - Optimising Outcomes - A framework for all staff working with people with Autism Spectrum Disorders, their families and carers. Edinburgh, 2014. Available:

Source: PubMed

Sponsors et collaborateurs

Les conditions médicales

Interventions en matière de drogue

3
S'abonner