Abstract
Many autistic children access some form of early intervention, but little is known about the value for money of different programs. We completed a scoping review of full economic evaluations of early interventions for autistic children and/or their families. We identified nine studies and reviewed their methods and quality. Most studies involved behavioral interventions. Two were trial-based, and the others used various modelling methods. Clinical measures were often used to infer dependency levels and quality-adjusted life-years. No family-based or negative outcomes were included. Authors acknowledged uncertain treatment effects. We conclude that economic evaluations in this field are sparse, methods vary, and quality is sometimes poor. Economic research is needed alongside longer-term clinical trials, and outcome measurement in this population requires further exploration.




Similar content being viewed by others
References
Aromataris, E., & Munn, Z. (2020). JBI Manual for Evidence Synthesis. JBI.
Buescher, A. V., Cidav, Z., Knapp, M., & Mandell, D. S. (2014). Costs of autism spectrum disorders in the United Kingdom and the United States. JAMA Pediatrics, 168(8), 721–728. https://doi.org/10.1001/jamapediatrics.2014.210
Byford, S., Cary, M., Barrett, B., Aldred, C. R., Charman, T., Howlin, P., Hudry, K., Leadbitter, K., Le Couteur, A., McConachie, H., Pickles, A., Slonims, V., Temple, K. J., Green, J., & Consortium, P. (2015). Cost-effectiveness analysis of a communication-focused therapy for pre-school children with autism: results from a randomised controlled trial. BMC Psychiatry, 15, 316. https://doi.org/10.1186/s12888-015-0700-x
Dallman, A. R., Artis, J., Watson, L., & Wright, S. (2021). Systematic review of disparities and differences in the access and use of allied health services amongst children with autism spectrum disorders. Journal of Autism and Developmental Disorders, 51(4), 1316–1330. https://doi.org/10.1007/s10803-020-04608-y
Dawson, G., Franz, L., & Brandsen, S. (2022). At a crossroads-reconsidering the goals of autism early behavioral intervention from a neurodiversity perspective. JAMA Pediatrics, 176(9), 839–840. https://doi.org/10.1001/jamapediatrics.2022.2299
DeVita-Raeburn, E., & Spectrum. (2016). Is the most common therapy for autism cruel? The Atlantic. Retrieved 22.3.22, from https://www.theatlantic.com/health/archive/2016/08/aba-autism-controversy/495272/
Drummond, M. F., Sculpher, M. J., Claxton, K., Stoddart, G. L., & Torrance, G. W. (2015). Methods for the economic evaluation of health care programmes. Oxford University Press.
Evers, S., Goossens, M., de Vet, H., van Tulder, M., & Ament, A. (2005). Criteria list for assessment of methodological quality of economic evaluations: Consensus on health economic criteria. International Journal of Technology Assessment in Health Care, 21(2), 240–245. https://doi.org/10.1017/s0266462305050324
Feeny, D., Furlong, W., Torrance, G. W., Goldsmith, C. H., Zhu, Z., Depauw, S., Denton, M., & Boyle, M. (2002). Multiattribute and single-attribute utility functions for the health utilities index mark 3 system. Medical Care. https://doi.org/10.1097/00005650-200202000-00006
Husereau, D., Drummond, M., Petrou, S., Carswell, C., Moher, D., Greenberg, D., Augustovski, F., Briggs, A. H., Mauskopf, J., Loder, E., & Force, C. T. (2013). Consolidated health economic evaluation reporting standards (CHEERS) statement. BMJ. https://doi.org/10.1136/bmj.f1049
Kislenko, C. (Apr 20, 2022). Why the 'treatment' of autism is a form of conversion therapy. Xtra*. https://xtramagazine.com/health/autism-conversion-therapy-221581
Knapp, M., Romeo, R., & Beecham, J. (2009). Economic cost of autism in the UK. Autism, 13(3), 317–336. https://doi.org/10.1177/1362361309104246
Kuhlthau, K., Orlich, F., Hall, T. A., Sikora, D., Kovacs, E. A., Delahaye, J., & Clemons, T. E. (2010). Health-related quality of life in children with autism spectrum disorders: Results from the autism treatment network. Journal of Autism and Developmental Disorders, 40(6), 721–729. https://doi.org/10.1007/s10803-009-0921-2
Lamsal, R., & Zwicker, J. D. (2017). Economic evaluation of interventions for children with neurodevelopmental disorders: opportunities and challenges. Applied Health Economics and Health Policy, 15(6), 763–772. https://doi.org/10.1007/s40258-017-0343-9
Lavelle, T. A., D’Cruz, B. N., Mohit, B., Ungar, W. J., Prosser, L. A., Tsiplova, K., Vera-Llonch, M., & Lin, P.-J. (2019). Family spillover effects in pediatric cost-utility analyses. Applied Health Economics and Health Policy, 17(2), 163–174. https://doi.org/10.1007/s40258-018-0436-0
Leadbitter, K., Aldred, C., McConachie, H., Le Couteur, A., Kapadia, D., Charman, T., Macdonald, W., Salomone, E., Emsley, R., & Green, J. (2018). The autism family experience questionnaire (AFEQ): An ecologically-valid, parent-nominated measure of family experience, quality of life and prioritised outcomes for early intervention. Journal of Autism and Developmental Disorders, 48(4), 1052–1062. https://doi.org/10.1007/s10803-017-3350-7
Leadbitter, K., Buckle, K. L., Ellis, C., & Dekker, M. (2021). Autistic self-advocacy and the neurodiversity movement: Implications for autism early intervention research and practice. Frontiers in Psychology. https://doi.org/10.3389/fpsyg.2021.635690
Leaf, J. B., Cihon, J. H., Leaf, R., McEachin, J., Liu, N., Russell, N., Unumb, L., Shapiro, S., & Khosrowshahi, D. (2022). Concerns about ABA-based intervention: An evaluation and recommendations. Journal of Autism and Developmental Disorders, 52(6), 2838–2853. https://doi.org/10.1007/s10803-021-05137-y
Lichtlé, J., Lamore, K., Pedoux, A., Downes, N., Mottron, L., & Cappe, E. (2022). Searching for what really matters: A thematic analysis of quality of life among preschool children on the autism spectrum. Journal of Autism and Developmental Disorders, 52, 2098–2111. https://doi.org/10.1007/s10803-021-05097-3
Lindgren, S., Wacker, D., Suess, A., Schieltz, K., Pelzel, K., Kopelman, T., Lee, J., Romani, P., & Waldron, D. (2016). Telehealth and autism: Treating challenging behavior at lower cost. Pediatrics, 137(Suppl 2), S167-175. https://doi.org/10.1542/peds.2015-2851O
Ludwig, J., & Phillips, D. A. (2008). Long-term effects of head start on low-income children. Annals of the New York Academy of Sciences, 1136(1), 257–268. https://doi.org/10.1196/annals.1425.005
Maenner, M. J., Shaw, K. A., Baio, J., Washington, A., Patrick, M., DiRienzo, M., Christensen, D. L., Wiggins, L. D., Pettygrove, S., Andrews, J. G., Lopez, M., Hudson, A., Baroud, T., Schwenk, Y., White, E., Rosenberg, C. R., Lee, L.-C., Harrington, R. A., Huston, M., . . . Dietz, P. M. (2020). Prevalence of Autism Spectrum Disorder Among Children Aged 8 Years — Autism and Developmental Disabilities Monitoring Network, 11 Sites, United States, 2016. Atlanta: Centers for Disease Control and Prevention (CDC)
Magiati, I., Tay, X. W., & Howlin, P. (2014). Cognitive, language, social and behavioural outcomes in adults with autism spectrum disorders: A systematic review of longitudinal follow-up studies in adulthood. Clinical Psychology Review, 34(1), 73–86. https://doi.org/10.1016/j.cpr.2013.11.002
May, T., Brignell, A., & Williams, K. (2020). Autism spectrum disorder prevalence in children aged 12–13 years from the longitudinal study of Australian children. Autism Research, 13(5), 821–827. https://doi.org/10.1002/aur.2286
McGowan, J., Sampson, M., Salzwedel, D. M., Cogo, E., Foerster, V., & Lefebvre, C. (2016). PRESS peer review of electronic search strategies: 2015 guideline statement. Journal of Clinical Epidemiology, 75, 40–46. https://doi.org/10.1016/j.jclinepi.2016.01.021
Motiwala, S. S., Gupta, S., Lilly, M. B., Ungar, W. J., & Coyte, P. C. (2006). The cost-effectiveness of expanding intensive behavioural intervention to all autistic children in Ontario. Healthcare Policy, 1, 135–151.
Munn, Z., Peters, M. D. J., Stern, C., Tufanaru, C., McArthur, A., & Aromataris, E. (2018). Systematic review or scoping review? Guidance for authors when choosing between a systematic or scoping review approach. BMC Medical Research Methodology, 18(1), 143. https://doi.org/10.1186/s12874-018-0611-x
National Institute for Health and Care Excellence. (2021). Developing NICE guidelines: the manual (PMG20)
National Disability Insurance Agency. (2022). Participant numbers and plan budget data https://data.ndis.gov.au/data-downloads#budget
Odnoletkova, I., Goderis, G., Pil, L., Nobels, F., Aertgeerts, B., Annemans, L., & Ramaekers, D. (2014). Cost-effectiveness of therapeutic education to prevent the development and progression of type 2 diabetes: Systematic review. Journal of Diabetes & Metabolism. https://doi.org/10.4172/2155-6156.1000438
Page, M. J., McKenzie, J. E., Bossuyt, P. M., Boutron, I., Hoffmann, T. C., Mulrow, C. D., Shamseer, L., Tetzlaff, J. M., Akl, E. A., Brennan, S. E., Chou, R., Glanville, J., Grimshaw, J. M., Hróbjartsson, A., Lalu, M. M., Li, T., Loder, E. W., Mayo-Wilson, E., McDonald, S., & Moher, D. (2021). The PRISMA 2020 statement: An updated guideline for reporting systematic reviews. BMJ. https://doi.org/10.1136/bmj.n71
Parker, S. (21 Mar 2015). Autism: does ABA therapy open society's doors to children, or impose conformity? The Guardian. https://www.theguardian.com/society/2015/mar/20/autism-does-aba-therapy-open-societys-doors-to-children-or-impose-conformity
Payakachat, N., Tilford, J. M., Kuhlthau, K. A., van Exel, N. J., Kovacs, E., Bellando, J., Pyne, J. M., & Brouwer, W. B. (2014). Predicting health utilities for children with autism spectrum disorders. Autism Research, 7(6), 649–663. https://doi.org/10.1002/aur.1409
Penner, M., Rayar, M., Bashir, N., Roberts, S. W., Hancock-Howard, R. L., & Coyte, P. C. (2015). Cost-effectiveness analysis comparing pre-diagnosis autism spectrum disorder (ASD)-targeted intervention with ontario’s autism intervention program. Journal of Autism and Developmental Disorders, 45(9), 2833–2847. https://doi.org/10.1007/s10803-015-2447-0
Peters, M. D. J., Marnie, C., Tricco, A. C., Pollock, D., Munn, Z., Alexander, L., McInerney, P., Godfrey, C. M., & Khalil, H. (2020). Updated methodological guidance for the conduct of scoping reviews. JBI. https://doi.org/10.11124/JBIES-20-00167
Philips, Z., Bojke, L., Sculpher, M., Claxton, K., & Golder, S. (2006). Good practice guidelines for decision-analytic modelling in health technology assessment: A review and consolidation of quality assessment. PharmacoEconomics, 24, 355–371.
Piccininni, C., Bisnaire, L., & Penner, M. (2017). Cost-effectiveness of wait time reduction for intensive behavioral intervention services in Ontario Canada. JAMA Pediatrics, 171(1), 23–30. https://doi.org/10.1001/jamapediatrics.2016.2695
Prior, M., & Roberts, J. (2006). Early Intervention for Children with Autism Spectrum Disorders: Guidelines for Best Practice.
Pye, K., Jackson, H., Iacono, T., & Shiell, A. (2021). Early intervention for young children with autism spectrum disorder: protocol for a scoping review of economic evaluations. Systematic Reviews 10(1). https://doi.org/10.1186/s13643-021-01847-7
Ramsey, S. D., Willke, R. J., Glick, H., Reed, S. D., Augustovski, F., Jonsson, B., Briggs, A., & Sullivan, S. D. (2015). Cost-effectiveness analysis alongside clinical trials II—An ISPOR good research practices task force report. Value in Health, 18(2), 161–172. https://doi.org/10.1016/j.jval.2015.02.001
Robins, D. L., Fein, D., Barton, M. L., & Green, J. A. (2001). The modifield checklist for autism in toddlers: An initial study investigating the early detection of autism and pervasive developmental disorders. Journal of Autism and Developmental Disorders, 31(2), 131–144. https://doi.org/10.1023/a:1010738829569
Rodgers, M., Marshall, D., Simmonds, M., Le Couteur, A., Biswas, M., Wright, K., Rai, D., Palmer, S., Stewart, L., & Hodgson, R. (2020). Interventions based on early intensive applied behaviour analysis for autistic children: A systematic review and cost-effectiveness analysis. Health Technology Assessment, 24(35), 1–306. https://doi.org/10.3310/hta24350
Sampaio, F., Feldman, I., Lavelle, T. A., & Skokauskas, N. (2021). The cost-effectiveness of treatments for attention deficit-hyperactivity disorder and autism spectrum disorder in children and adolescents: A systematic review. European Child & Adolescent Psychiatry. https://doi.org/10.1007/s00787-021-01748-z
Sandbank, M., Bottema-Beutel, K., Crowley, S., Cassidy, M., Dunham, K., Feldman, J. I., Crank, J., Albarran, S. A., Raj, S., Mahbub, P., & Woynaroski, T. G. (2020). Project AIM: Autism intervention meta-analysis for studies of young children. Psychological Bulletin, 146(1), 1–29. https://doi.org/10.1037/bul0000215
Sinclair, J. (1993). Don't mourn for us. Our Voice, 1(3). https://www.autreat.com/dont_mourn.html
Synergies Economic Consulting. (2013). Cost-benefit analysis of providing early intervention to children with autism. Retrieved 2020 Oct 01 from https://www.synergies.com.au/reports/cost-benefit-analysis-of-providing-early-intervention-to-children-with-autism/
Trembath, D., Varcin, K., Waddington, H., Sulek, R., Bent, C., Ashburner, J., Eapen, V., Goodall, E., Hudry, K., Roberts, J., Silove, N., & Whitehouse, A. (2022). Non-pharmacological interventions for autistic children: An umbrella review. Autism. https://doi.org/10.1177/13623613221119368
Tricco, A. C., Lillie, E., Zarin, W., O’Brien, K. K., Colquhoun, H., Levac, D., Moher, D., Peters, M. D. J., Horsley, T., Weeks, L., Hempel, S., Akl, E. A., Chang, C., McGowan, J., Stewart, L., Hartling, L., Aldcroft, A., Wilson, M. G., Garritty, C., & Straus, S. E. (2018). PRISMA extension for scoping reviews (PRISMA-ScR): checklist and explanation. Annals of Internal Medicine, 169(7), 467–473. https://doi.org/10.7326/M18-0850
Ungar, W. J. (2011). Challenges in health state valuation in paediatric economic evaluation. PharmacoEconomics, 29(8), 641–652. https://doi.org/10.2165/11591570-000000000-00000
United Nations Convention on the rights of persons with disabilities, (December 13, 2006). https://www.un.org/development/desa/disabilities/convention-on-the-rights-of-persons-with-disabilities.html
van Mastrigt, G. A., Hiligsmann, M., Arts, J. J., Broos, P. H., Kleijnen, J., Evers, S. M., & Majoie, M. H. (2016). How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: A five-step approach (part 1/3). Expert Review of Pharmacoeconomics & Outcomes Research, 16(6), 689–704. https://doi.org/10.1080/14737167.2016.1246960
Veritas Health Innovation. (2021). Covidence systematic review software. Melbourne, Australia. Available at www.covidence.org
Weinmann, S., Schwarzbach, C., Begemann, M., Roll, S., Vauth, C., Willich, S. N., & Greiner, W. (2009). Behavioural and skill-based early interventions in children with autism spectrum disorders. GMS Health Technology Assessment. https://doi.org/10.3205/hta000072
Whitehouse, A. J. O., Varcin, K., Waddington, H., Sulek, R., Bent, C., Ashburner, J., Eapen, V., Goodall, E., Hudry, K., Roberts, J., Silove, N., & Trembath, D. (2020). Interventions for children on the autism spectrum: A synthesis of research evidence. Brisbane: Autism CRC
Wijnen, B., Van Mastrigt, G., Redekop, W. K., Majoie, H., De Kinderen, R., & Evers, S. (2016). How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: Data extraction, risk of bias, and transferability (part 3/3). Expert Review of Pharmacoeconomics & Outcomes Research, 16(6), 723–732. https://doi.org/10.1080/14737167.2016.1246961
Williamson, I. O., Elison, J. T., Wolff, J. J., & Runge, C. F. (2020). Cost-effectiveness of MRI-based identification of presymptomatic autism in a high-risk population. Front Psychiatry, 11, 60. https://doi.org/10.3389/fpsyt.2020.00060
World Health Organization. (2001). International classification of functioning, disability, and health : ICF. In. Geneva: World Health Organization.
Yuen, T., Carter, M. T., Szatmari, P., & Ungar, W. J. (2018). Cost-effectiveness of universal or high-risk screening compared to surveillance monitoring in autism spectrum disorder. Journal of Autism and Developmental Disorders, 48(9), 2968–2979. https://doi.org/10.1007/s10803-018-3571-4
Acknowledgments
The authors gratefully acknowledge the contribution of Hannah Buttery (Senior Officer, Research Data Outputs) and Natalie Young (Research Advisor) of La Trobe University, in their support reviewing the search strategy.
Funding
This review was completed as part of a PhD candidature supported by the Australian Government’s Research Training Program.
Author information
Authors and Affiliations
Contributions
KP, AS and TI developed the concepts of this study. KP developed the search strategy with input from the other authors. KP and HJ completed screening and data extraction, with AS as third reviewer when required. KP drafted and finalised this paper with valuable contributions from the other authors, all of whom approved the final manuscript.
Corresponding author
Ethics declarations
Conflict of interest
The authors declare they have no competing interests.
Ethical Approval
Not applicable.
Consent to Participate
Not applicable.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Supplementary Information
Below is the link to the electronic supplementary material.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Pye, K., Jackson, H., Iacono, T. et al. Economic Evaluation of Early Interventions for Autistic Children: A Scoping Review. J Autism Dev Disord (2023). https://doi.org/10.1007/s10803-023-05938-3
Accepted:
Published:
DOI: https://doi.org/10.1007/s10803-023-05938-3

