Abstract
This study measured resource utilization and costs for pre-school autism spectrum disorder (ASD)-related services in community-based sectors from multiple payer perspectives in two Canadian provinces, Nova Scotia (NS) and New Brunswick (NB), during the 12 months prior to and following the start of early intensive behavioural intervention (EIBI). The results indicate significant differences between NB and NS in utilization of services and costs to families, public sector and society. Differences can be attributed to variation in EIBI delivery models and may also be influenced by differences in diagnostic assessment practices. The study results provide resource utilization rates and costs which could be used in future economic evaluations and to inform policy making to improve outcomes for children with ASD.
Similar content being viewed by others
References
American Psychiatric Association. (2013). Diagnostic and statistical manual of mental disorders (5th ed.). Arlington: American Psychiatric Publishing.
Anagnostou, E., Zwaigenbaum, L., Szatmari, P., Fombonne, E., Fernandez, B. A., Woodbury-Smith, M., et al. (2014). Autism spectrum disorder: Advances in evidence-based practice. Canadian Medical Association Journal, 186(7), 509–519.
Auditor General of Ontario (2013). 2013 Annual report of the office of the Auditor General of Ontario: 3.01 Autism services and supports for children. Retrieved July 30, 2018, from http://www.auditor.on.ca/en/content/annualreports/arreports/en13/2013ar_en_web.pdf.
Auditor General of Ontario (2015). 2015 Annual report of the office of the Auditor General of Ontario: Autism services and supports for children, follow-up to VFM Sect. 3.01, 2013 Annual Report. Retrieved July 30, 2018, from http://www.auditor.on.ca/en/content/annualreports/arreports/en15/2015AR_en_final.pdf.
Australian Government (2016). Disability and carers. What help is available? Retrieved June 8, 2018, from https://www.dss.gov.au/disability-and-carers/programmes-services/for-people-with-disability/what-help-is-available#1.
Bank of Canada (2017). Annual Exchange Rates. Retrieved March 21, 2018, from https://www.bankofcanada.ca/rates/exchange/annual-average-exchange-rates/.
Barrett, B., Byford, S., Sharac, J., Hudry, K., Leadbitter, K., Temple, K., et al. (2012). Service and wider societal costs of very young children with autism in the UK. Journal of Autism and Developmental Disorders, 42(5), 797–804. https://doi.org/10.1007/s10803-011-1306-x.
Bryson, S. E., Koegel, L. K., Koegel, R. L., Openden, D., Smith, I. M., & Nefdt, N. (2007). Large scale dissemination and community implementation of pivotal response treatment: Program description and preliminary data. Research & Practice for Persons with Severe Disabilities, 32(2), 142–153.
Buescher, A. S., 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.
Chasson, G. S., Harris, G. E., & Neely, W. J. (2007). Cost comparison of early intensive behavioral intervention and special education for children with autism. Journal of Child and Family Studies, 16(3), 401–413. https://doi.org/10.1007/s10826-006-9094-1.
Christensen, D. L., Baio, J., Braun, K. V. N., Bilder, D., Charles, J., Constantino, J. N., et al. (2016). Prevalence and characteristics of autism spectrum disorder among children aged 8 years—Autism and Developmental Disabilities Monitoring Network, 11 Sites, United States, 2012. MMWR Surveillance Summaries, 65(-3), 1–23. https://doi.org/10.15585/mmwr.ss6503a1.
Cidav, Z., Lawer, L., Marcus, S. C., & Mandell, D. S. (2013). Age-related variation in health service use and associated expenditures among children with autism. Journal of Autism and Developmental Disorders, 43(4), 924–931. https://doi.org/10.1007/s10803-012-1637-2.
Cohen, H., Amerine-Dickens, M., & Smith, T. (2006). Early intensive behavioral treatment: replication of the UCLA model in a community setting. Journal of Developmental & Behavioral Pediatrics, 27(2), S145–S155.
Dudley, C., & Zwicker, J. (2014). The value of caregiver time: Costs of support and care for individuals living with austism spectrum disorder. In: SPP Research Papers. Calgary: The School of Public Policy, University of Calgary.
Eikeseth, S., Smith, T., Jahr, E., & Eldevik, S. (2007). Outcome for children with autism who began intensive behavioral treatment between ages 4 and 7: A comparison controlled study. Behavior Modification, 31(3), 264–278. https://doi.org/10.1177/0145445506291396.
Fitzmaurice, G. M., Laird, N., & Ware, J. (2011). Applied longitudinal analysis (2nd ed). Hoboken: Wiley.
Ganz, M. L. (2007). The lifetime distribution of the incremental societal costs of autism. Archives of Pediatrics & Adolescent Medicine, 161(4), 343–349. https://doi.org/10.1001/archpedi.161.4.343.
Government of British Columbia (2018). The Autism Funding Program. Retrieved November 30, 2018, from http://autisminfo.gov.bc.ca/autism-funding-program/.
Harris, P. A., Taylor, R., Thielke, R., Payne, J., Gonzalez, N., & Conde, J. G. (2009). Research Electronic Data Capture (REDCap) - A metadata-driven methodology and workflow process for providing translational research informatics support. Journal of biomedical informatics, 42(2), 377–381. https://doi.org/10.1016/j.jbi.2008.08.010.
Hendry, L. (2016). Quebec families turn to private autism therapies—at staggering cost. Retrieved from http://www.cbc.ca/news/canada/montreal/autism-services-inadequate-1.3589974.
Horlin, C., Falkmer, M., Parsons, R., Albrecht, M. A., & Falkmer, T. (2014). The cost of autism spectrum disorders. PLoS ONE. 9(9), e106552.
Janet, A. T., Gary, K. G., & Richard, H. J. (2002). Analysis of repeated measures data with clumping at zero. Statistical Methods in Medical Research, 11(4), 341–355. https://doi.org/10.1191/0962280202sm291ra.
Järbrink, K., Fombonne, E., & Knapp, M. (2003). Measuring the parental, service and cost impacts of children with autistic spectrum disorder: A pilot study. Journal of Autism and Developmental Disorders, 33(4), 395–402. https://doi.org/10.1023/a:1025058711465.
Keenan, M., Dillenburger, K., Röttgers, H. R., Dounavi, K., Jónsdóttir, S. L., Moderato, P., et al. (2015). Autism and ABA: The gulf between North America and Europe. Review Journal of Autism and Developmental Disorders, 2(2), 167–183. https://doi.org/10.1007/s40489-014-0045-2.
Koegel, R., & Koegel, L. (2006). Pivotal response treatments for autism. Baltimore: Brookes Publishing.
Koegel, R., Koegel, L., Vernon, T., & Brookman-Frazee, L. (2014). Pivotal response treatment for individuals with autism spectrum disorder. In J. R. Weisz & A. E. Kazdin (Eds.), Evidence-Based psychotherapies for children and adolescents. New York: Guilford Publications.
Koegel, R. L., O’Dell, M., & Koegel, L. K. (1987). A natural language teaching paradigm for nonverbal autistic children. Journal of Autism and Developmental Disorders, 17(2), 187–200. https://doi.org/10.1007/bf01495055.
L & M Policy Research. (2014). Autism spectrum disorders (ASD): State of the states of services and supports for people with ASD. Washington, DC: Centers for Medicare and Medicaid Services.
Lai, J. K. Y., & Weiss, J. A. (2017). Priority service needs and receipt across the lifespan for individuals with autism spectrum disorder. Autism Research, 10(8), 1436–1447. https://doi.org/10.1002/aur.1786.
Lavelle, T. A., Weinstein, M. C., Newhouse, J. P., Munir, K., Kuhlthau, K. A., & Prosser, L. A. (2014). Economic burden of childhood autism spectrum disorders. Pediatrics, 133(3), e520–e529. https://doi.org/10.1542/peds.2013-0763.
Leigh, J. P., & Du, J. (2015). Brief report: Forecasting the economic burden of autism in 2015 and 2025 in the United States. Journal of Autism and Developmental Disorders, 45(12), 4135–4139. https://doi.org/10.1007/s10803-015-2521-7.
Liu, L., Cowen, M. E., Strawderman, R. L., & Shih, Y.-C. T. (2010). A flexible two-part random effects model for correlated medical costs. Journal of Health Economics, 29(1), 110–123. https://doi.org/10.1016/j.jhealeco.2009.11.010.
Lovaas, O. I. (1987). Behavioral treatment and normal educational and intellectual functioning in young autistic children. Journal of Consulting and Clinical Psychology, 55(1), 3–9. https://doi.org/10.1037/0022-006x.55.1.3.
Malehi, A. S., Pourmotahari, F., & Angali, K. A. (2015). Statistical models for the analysis of skewed healthcare cost data: A simulation study. Health Economics Review, 5, 11. https://doi.org/10.1186/s13561-015-0045-7.
Manning, W. G., Basu, A., & Mullahy, J. (2005). Generalized modeling approaches to risk adjustment of skewed outcomes data. Journal of Health Economics, 24(3), 465–488. https://doi.org/10.1016/j.jhealeco.2004.09.011.
Manning, W. G., & Mullahy, J. (2001). Estimating log models: To transform or not to transform? Journal of Health Economics, 20(4), 461–494. https://doi.org/10.1016/S0167-6296(01)00086-8.
McEachin, J., Smith, T., & Lovaas, O. I. (1993). Long-term outcome for children with autism who received early intensive behavioral treatment. American Journal on Mental Retardation, 97(4), 359.
Mihaylova, B., Briggs, A., O’Hagan, A., & Thompson, S. G. (2011). Review of statistical methods for analysing healthcare resources and costs. Health Economics, 20(8), 897–916. https://doi.org/10.1002/hec.1653.
Nixon, R. M., & Thompson, S. G. (2004). Parametric modelling of cost data in medical studies. Statistics in Medicine, 23(8), 1311–1331. https://doi.org/10.1002/sim.1744.
Ofner, M., Coles, A., Decou, M. L., Do, M. T., Bienek, A., Snider, J., et al. (2018). Autism spectrum disorder among children and youth in Canada 2018. Ottawa: Public Health Agency of Canada.
Ontario Ministry of Children and Youth Services (2018). Ontario autism program. Retrieved July 21, 2018, from http://www.children.gov.on.ca/htdocs/English/specialneeds/autism/ontario-autism-program.aspx.
Parkin, E., Bate, A., Long, R., Gheera, M., Powell, A., Bellis, A., et al. (2018). Autism – overview of UK policy and services. In Commons Briefing papers CBP-7172. London: The House of Commons Library.
Peacock, G., Amendah, D., Ouyang, L., & Grosse, S. D. (2012). Autism spectrum disorders and health care expenditures: The effects of co-occurring conditions. Journal of Developmental & Behavioral Pediatrics, 33(1), 2–8. https://doi.org/10.1097/DBP.0b013e31823969de.
Peters-Scheffer, N., Didden, R., Korzilius, H., & Matson, J. (2012). Cost comparison of early intensive behavioral intervention and treatment as usual for children with autism spectrum disorder in the Netherlands. Research in Developmental Disabilities, 33(6), 1763–1772. https://doi.org/10.1016/j.ridd.2012.04.006.
Prior, M., & Roberts, J. (2012). Early intervention for children with autism spectrum disorders: ‘Guidelines for good practice’ 2012. Retrieved June 8, 2018, from https://www.dss.gov.au/sites/default/files/documents/08_2014/6006_-_accessible_-_early_intervention_practice_guidelines_0.pdf.
R Core Team. (2016). R: A language and environment for statistical computing. Vienna: R Foundation for Statistical Computing.
Reichow, B., Hume, K., Barton, E. E., & Boyd, B. A. (2018). Early intensive behavioral intervention (EIBI) for young children with autism spectrum disorders (ASD). Cochrane Database of Systematic Reviews. https://doi.org/10.1002/14651858.CD009260.pub3.
Reichow, B., & Wolery, M. (2008). Comprehensive synthesis of Early Intensive Behavioral Interventions for young children with autism based on the UCLA Young Autism Project model. Journal of Autism and Developmental Disorders, 39(1), 23. https://doi.org/10.1007/s10803-008-0596-0.
Sallows, G. O., & Graupner, T. D. (2005). Intensive Behavioral treatment for children with autism: four-year outcome and predictors. American Journal on Mental Retardation, 110(6), 417–438. https://doi.org/10.1352/0895-8017(2005)110%5B417:IBTFCW%5D2.0.CO;2.
Salomone, E., Beranová, Š, Bonnet-Brilhault, F., Briciet Lauritsen, M., Budisteanu, M., Buitelaar, J., et al. (2015). Use of early intervention for young children with autism spectrum disorder across Europe. Autism, 20(2), 233–249. https://doi.org/10.1177/1362361315577218.
Shepherd, C. A., & Waddell, C. (2015). A qualitative study of autism policy in Canada: Seeking consensus on children’s services. Journal of Autism and Developmental Disorders, 45(11), 3550–3564. https://doi.org/10.1007/s10803-015-2502-x.
Simon, G. T., & Richard, M. N. (2005). How sensitive are cost-effectiveness analyses to choice of parametric distributions? Medical Decision Making, 25(4), 416–423. https://doi.org/10.1177/0272989X05276862.
Smith, I. M., Flanagan, H. E., Garon, N., & Bryson, S. E. (2015). Effectiveness of community-based early intervention based on Pivotal Response Treatment. Journal of Autism and Developmental Disorders, 45(6), 1858–1872. https://doi.org/10.1007/s10803-014-2345-x.
Smith, I. M., Flanagan, H. E., Ungar, W. J., D.’Entremont, Garon, B., den Otter, N., et al. (2019). Comparing the 1-year impact of preschool autism intervention programs in two Canadian provinces. Autism Research. https://doi.org/10.1002/aur.2072.
Smith, I. M., Koegel, R. L., Koegel, L. K., Openden, D. A., Accardi, C., Fossum, K. L., et al. (2010). Effectiveness of a novel community-based early intervention model for children with autistic spectrum disorder. American Journal on Intellectual and Developmental Disabilities, 115(6), 504–523. https://doi.org/10.1352/1944-7558-115.6.504.
Smith, T., Groen, A., & Wynn, J. (2000). Randomized trial of intensive early intervention for children with pervasive developmental disorder. American Journal on Mental Retardation. 105(4), 269–285, https://doi.org/10.1352/0895-8017(2000)105%3C0269:rtoiei%3E2.0.co;2.
Statistics Canada (2017). Table 14-10-0206-01 Average hourly earnings for employees paid by the hour, by industry, annual. Retrieved July 31, 2018, from https://www150.statcan.gc.ca/t1/tbl1/en/tv.action?pid=1410020601.
Statistics Canada (2018). Table 18-10-0005-01 Consumer Price Index, annual average, not seasonally adjusted. Retrieved July 31, 2018, from https://www150.statcan.gc.ca/t1/tbl1/en/tv.action?pid=1810000501.
Ungar, W. J., Tsiplova, K., Millar, N., & Smith, I. M. (2018). Development of the Resource Use Questionnaire (RUQ-P) for families with preschool children with neurodevelopmental disorders: Validation in children with autism spectrum disorder. Clinical Practice in Pediatric Psychology, 6(2), 164–178. https://doi.org/10.1037/cpp0000226.
Vohra, R., Madhavan, S., Sambamoorthi, U., & St Peter, C. (2014). Access to services, quality of care, and family impact for children with autism, other developmental disabilities, and other mental health conditions. Autism, 18(7), 815–826. https://doi.org/10.1177/1362361313512902.
Volden, J., Duku, E., Shepherd, C., Georgiades, S., Bennett, T., et al. (2015). Service utilization in a sample of preschool children with autism spectrum disorder: A Canadian snapshot. Paediatr Child Health, 20(8), e43–e47.
Yingling, M. E., Hock, R. M., & Bell, B. A. (2018). Time-lag between diagnosis of autism spectrum disorder and onset of publicly-funded Early Intensive Behavioral Intervention: Do race-ethnicity and neighborhood matter? Journal of Autism and Developmental Disorders, 48(2), 561–571. https://doi.org/10.1007/s10803-017-3354-3.
Zhou, X.-H., Stroupe, K. T., & Tierney, W. M. (2001). Regression analysis of health care charges with heteroscedasticity. Journal of the Royal Statistical Society: Series C (Applied Statistics), 50(3), 303–312. https://doi.org/10.1111/1467-9876.00235.
Acknowledgments
This study was funded by a Canadian Institutes of Health Research ‘Partnerships for Health Systems Improvement’ operating Grant (#PHE 122189), by partner funding from the Nova Scotia Health Research Foundation and New Brunswick Health Research Foundation, and by the 2017 SickKids Lunenfeld Summer Studentship program. PATI study’s Principal Investigator Smith was supported by the Joan and Jack Craig Chair in Autism Research. We thank Noreen Millar, Jennifer Fox, Susan Harvey, and Marie-Lyn Fackenthall, research coordinators, for their help with collecting and using the PATI study data. We acknowledge the work of Salar Sadri on resource pricing and costing. We also appreciate the help of PATI team members, service providers and Nova Scotia EIBI managers who provided cost data. We would especially like to thank all of the participating families.
Author information
Authors and Affiliations
Contributions
KT participated in the interpretation of the data, performed statistical analysis and drafted the manuscript; WU conceived the study, contributed to its design and coordination and to the interpretation and statistical analysis of the data and drafted the manuscript; BD, HF, NG, JD, CW and PM participated in the design and coordination of the study and the interpretation of the data; NL contributed to the design and coordination of the study; SB conceived of the study and participated in its design; IS conceived of the study, participated in its design and coordination and contributed to the interpretation of the data. All authors read and approved the final manuscript.
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical Approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed Consent
Informed consent was obtained from all individual participants included in the study.
Additional information
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Patricia Murray—now retired.
Electronic supplementary material
Below is the link to the electronic supplementary material.
Rights and permissions
About this article
Cite this article
Tsiplova, K., Ungar, W.J., Flanagan, H.E. et al. Types of Services and Costs of Programs for Preschoolers with Autism Spectrum Disorder Across Sectors: A Comparison of Two Canadian Provinces. J Autism Dev Disord 49, 2492–2508 (2019). https://doi.org/10.1007/s10803-019-03993-3
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10803-019-03993-3