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Cost-Effectiveness of Universal or High-Risk Screening Compared to Surveillance Monitoring in Autism Spectrum Disorder

  • Tracy Yuen
  • Melissa T. Carter
  • Peter Szatmari
  • Wendy J. Ungar
Original Paper

Abstract

The American Academy of Pediatrics recommends universal screening for autism spectrum disorder at 18 and 24 months. This study compared the cost-effectiveness of universal or high-risk screening to surveillance monitoring. Simulation models estimated the costs and outcomes from birth to age 6 years. The incremental cost per child diagnosed by 36 months was $41,651.6 for high-risk screening and $757,116.9 for universal screening from the societal perspective. Universal screening may not be a cost-effective approach to increase earlier treatment initiation, as most children initiated treatment after age 60 months. Eliminating wait times resulted in more children initiated treatment by 48 months, but at a high initial cost that may be offset by future cost-savings related to better outcomes.

Keywords

Autism spectrum disorder Cost-effectiveness analysis Health services research Screening 

Notes

Acknowldgements

Tracy Yuen was supported through the Canada Institutes of Health Research Autism Research Training Program, Doctoral Autism Scholars Award, Ontario Graduate Scholarship and RestraComp Hospital for Sick Children Foundation Student Scholarship Program. The funders had no role in design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication. The work is based on the doctoral dissertation of Dr. T. Yuen. The authors wish to thank Drs. Jessica Brian, Susan Bryson, Isabel Smith and Lonnie Zwaigenbaum for sharing data from the “Canadian high risk infant cohort” (Infant Sibling study) which informed the observed ASD status for all children and trajectories for high-risk children.

Author Contributions

All authors (TY, MTC, PS, WJU) contributed to the conceptualization of the model, interpretation of findings, editing of the manuscript andn provided final approval of the paper. TY and WJU were responsible for model building, data analysis and drafting of the manuscript.

Compliance with Ethical Standards

Conflict of interest

All authors declare no conflicts of interest.

Supplementary material

10803_2018_3571_MOESM1_ESM.docx (77 kb)
Supplementary material 1 (DOCX 77 KB)

References

  1. Al-Qabandi, M., Gorter, J. W., & Rosenbaum, P. (2011). Early autism detection: Are we ready for routine screening? Pediatrics, 128(1), e211–e217.  https://doi.org/10.1542/peds.2010-1881.CrossRefPubMedGoogle Scholar
  2. American Academy of Pediatrics. (2015). Recommendations for preventive pediatric health care—Periodicity schedule. https://www.aap.org/en-us/Documents/PeriodicitySchedule2015_Visionscreening.pdf.
  3. Auditor General of Ontario. (2013). Autism services and supports for children. Office of Auditor General of Ontario.Google Scholar
  4. Auditor General of Ontario. (2015). Autism services and supports for children. Office of Auditor General of Ontario.Google Scholar
  5. Begeer, S., Bouk, S. E., Boussaid, W., Terwogt, M. M., & Koot, H. M. (2009). Underdiagnosis and referral bias of autism in ethnic minorities. Journal of Autism and Developmental Disorders, 39(1), 142–148.  https://doi.org/10.1007/s10803-008-0611-5.CrossRefPubMedGoogle Scholar
  6. British Columbia Psychological Association. (2014). Suggested current market rate 2014–2015. British Columbia Psychological Association.Google Scholar
  7. Buescher, A. V. 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.  https://doi.org/10.1001/jamapediatrics.2014.210.CrossRefPubMedGoogle Scholar
  8. Centers for Disease Control and Prevention. (2015). Developmental milestone checklist. Division of Birth Defects, National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention. Retrieved January 21, 2015 from http://www.cdc.gov/ncbddd/actearly/milestones/.
  9. Centers for Disease Control and Prevention. (2016). Prevalence and characteristics of autism spectrum disorder among children aged 8 years- Autism and Developmental Disabilities Monitoring Network, 11 Sites, United States 2012. Morbidity and Mortality Weekly Report, 65(3), 1–21Google Scholar
  10. 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.CrossRefGoogle Scholar
  11. Chawarska, K., Klin, A., Paul, R., & Volkmar, F. (2007). Autism spectrum disorder in the second year: Stability and change in syndrome expression. Journal of Child Psychology and Psychiatry, 48(2), 128–138.  https://doi.org/10.1111/j.1469-7610.2006.01685.x.CrossRefPubMedGoogle Scholar
  12. Chlebowski, C., Robins, D. L., Barton, M. L., & Fein, D. (2013). Large-scale use of the modified checklist for autism in low-risk toddlers. Pediatrics, 131(4), e1121–e1127.  https://doi.org/10.1542/peds.2012-1525.CrossRefPubMedPubMedCentralGoogle Scholar
  13. 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.CrossRefPubMedPubMedCentralGoogle Scholar
  14. Cidav, Z., Munson, J., Estes, A., Dawson, G., Rogers, S., & Mandell, D. (2017). Cost offset associated with early start denver model for children with autism. Journal of the American Academy of Child and Adolescent Psychiatry, 56(9), 777–783.  https://doi.org/10.1016/j.jaac.2017.06.007.CrossRefPubMedGoogle Scholar
  15. Dawson, G. (2016). Why it’s important to continue universal autism screening while research fully examines its impact. JAMA Pediatrics, 170(6), 527.  https://doi.org/10.1001/jamapediatrics.2016.0163.CrossRefPubMedGoogle Scholar
  16. Fein, D. (2016). Commentary on USPSTF final statement on universal screening for autism. Journal of Developmental & Behavioral Pediatrics, 37(7), 573–578.  https://doi.org/10.1097/DBP.0000000000000345.CrossRefGoogle Scholar
  17. Filipek, P. A., Accardo, P. J., Ashwal, S., Baranek, G. T., Cook, E. H., Dawson, G., et al. (2000). Practice parameter: Screening and diagnosis of autism: Report of the Quality Standards Subcommittee of the American Academy of Neurology and the Child Neurology Society. Neurology, 55(4), 468–479.CrossRefPubMedGoogle Scholar
  18. Flores, G., & Lin, H. (2013). Trends in racial/ethnic disparities in medical and oral health, access to care, and use of services in US children: Has anything changed over the years? International Journal for Equity in Health, 12, 10.  https://doi.org/10.1186/1475-9276-12-10.CrossRefPubMedPubMedCentralGoogle Scholar
  19. Granpeesheh, D., Dixon, D. R., Tarbox, J., Kaplan, A. M., & Wilke, A. E. (2009). The effects of age and treatment intensity on behavioral intervention outcomes for children with autism spectrum disorders. Research in Autism Spectrum Disorders, 3(4), 1014–1022.  https://doi.org/10.1016/j.rasd.2009.06.007.CrossRefGoogle Scholar
  20. Grønborg, T. K., Schendel, D. E., & Parner, E. T. (2013). Recurrence of autism spectrum disorders in full- and half-siblings and trends over time: A population-based cohort study. JAMA Pediatrics, 167(10), 947.  https://doi.org/10.1001/jamapediatrics.2013.2259.CrossRefPubMedPubMedCentralGoogle Scholar
  21. Guerrero, A. D., Rodriguez, M. A., & Flores, G. (2011). Disparities in provider elicitation of parents’ developmental concerns for US children. Pediatrics, 128(5), 901–909.  https://doi.org/10.1542/peds.2011-0030.CrossRefPubMedGoogle Scholar
  22. Guevara, J. P., Gerdes, M., Localio, R., Huang, Y. V., Pinto-Martin, J., Minkovitz, C. S., et al. (2013). Effectiveness of developmental screening in an urban setting. Pediatrics, 131(1), 30–37.  https://doi.org/10.1542/peds.2012-0765.CrossRefPubMedGoogle Scholar
  23. Guthrie, W., Swineford, L. B., Nottke, C., & Wetherby, A. M. (2013). Early diagnosis of autism spectrum disorder: Stability and change in clinical diagnosis and symptom presentation. Journal of Child Psychology and Psychiatry, 54(5), 582–590.  https://doi.org/10.1111/jcpp.12008.CrossRefPubMedPubMedCentralGoogle Scholar
  24. Hagan, J. F., Shaw, S. M., & Duncan, P. (2008). Bright futures: Guidelines for health supervision of infants, children, and adolescents. Pocket guide (3rd edn.). Elk Grove Village, IL: American Academy of Pediatrics.Google Scholar
  25. Harris, S. L., & Handleman, J. S. (2000). Age and IQ at intake as predictors of placement for young children with autism: A four- to six-year follow-up. Journal of Autism and Developmental Disorders, 30(2), 137–142.CrossRefPubMedGoogle Scholar
  26. Huerta, M., Bishop, S. L., Duncan, A., Hus, V., & Lord, C. (2012). Application of DSM-5 criteria for autism spectrum disorder to three samples of children with DSM-IV diagnoses of pervasive developmental disorders. The American Journal of Psychiatry, 169(10), 1056–1064.  https://doi.org/10.1176/appi.ajp.2012.12020276.CrossRefPubMedGoogle Scholar
  27. Jacobson, J. W., Mulick, J. A., & Green, G. (1998). Cost-benefit estimates for early intensive behavioral intervention for young children with autism—general model and single state case. Behavioral Interventions, 13(4), 201–226 https://doi.org/10.1002/(SICI)1099-078X(199811)13:4%3C201::AID-BIN17%3E3.0.CO;2-R.CrossRefGoogle Scholar
  28. Johnson, C. P., Myers, S. M. & the Council on Children With Disabilities. (2007). Identification and evaluation of children with autism spectrum disorders. Pediatrics, 120(5), 1183–1215.  https://doi.org/10.1542/peds.2007-2361.CrossRefPubMedGoogle Scholar
  29. Kasari, C., Gulsrud, A., Freeman, S., Paparella, T., & Hellemann, G. (2012). Longitudinal follow-up of children with autism receiving targeted interventions on joint attention and play. Journal of the American Academy of Child & Adolescent Psychiatry, 51(5), 487–495.  https://doi.org/10.1016/j.jaac.2012.02.019.CrossRefGoogle Scholar
  30. Kasari, C., Gulsrud, A. C., Wong, C., Kwon, S., & Locke, J. (2010). Randomized controlled caregiver mediated joint engagement intervention for toddlers with autism. Journal of Autism and Developmental Disorders, 40(9), 1045–1056.  https://doi.org/10.1007/s10803-010-0955-5.CrossRefPubMedPubMedCentralGoogle Scholar
  31. Limperopoulos, C., Bassan, H., Sullivan, N. R., Soul, J. S., Robertson, R. L., Moore, M., et al. (2008). Positive screening for autism in ex-preterm infants: Prevalence and risk factors. Pediatrics, 121(4), 758–765.  https://doi.org/10.1542/peds.2007-2158.CrossRefPubMedPubMedCentralGoogle Scholar
  32. Liptak, G. S., Stuart, T., & Auinger, P. (2006). Health care utilization and expenditures for children with autism: Data from U.S. national samples. Journal of Autism and Developmental Disorders, 36(7), 871–879.  https://doi.org/10.1007/s10803-006-0119-9.CrossRefPubMedGoogle Scholar
  33. Mandell, D. S., & Mandy, W. (2015). Should all young children be screened for autism spectrum disorder? Autism, 19(8), 895–896.CrossRefPubMedGoogle Scholar
  34. Mandell, D. S., Novak, M. M., & Zubritsky, C. D. (2005). Factors associated with age of diagnosis among children with autism spectrum disorders. Pediatrics, 116(6), 1480–1486.  https://doi.org/10.1542/peds.2005-0185.CrossRefPubMedPubMedCentralGoogle Scholar
  35. MATLAB. (2017). SimEvent. Natick, MA: MathWorks Inc.Google Scholar
  36. McPartland, J. C., Reichow, B., & Volkmar, F. R. (2012). Sensitivity and specificity of proposed DSM-5 diagnostic criteria for autism spectrum disorder. Journal of the American Academy of Child & Adolescent Psychiatry, 51(4), 368–383.  https://doi.org/10.1016/j.jaac.2012.01.007.CrossRefGoogle Scholar
  37. Ministry of Health and Long-Term Care. (2015). Schedule of benefits for physician services under the Health Insurance Act. Retrieved November 9, 2015 from http://www.health.gov.on.ca/english/providers/program/ohip/sob/physserv/physserv_mn.html.
  38. Nachshen, J., Garcin, N., Moxness, K., Tremblay, Y., Hutchinson, P., Lachance, A., et al. (2008). Screening, assessment and diagnosis of autism spectrum disorder in young children: Canadian best practice guidelines. Montreal: Miriam Foundation.Google Scholar
  39. Ontario Association of Speech-Language Pathologists and Audiologists. (2016). Recommended fee schedule for speech language pathology services. Ontario Association of Speech-Language Pathologists and Audiologists.Google Scholar
  40. Ontario Psychological Association. (2015). Guidelines for fees and billing practices. Ontario: Ontario Psychological AssociationGoogle Scholar
  41. Ouellette-Kuntz, H. M., Coo, H., Lam, M., Breitenbach, M. M., Hennessey, P. E., Jackman, P. D., et al. (2014). The changing prevalence of autism in three regions of Canada. Journal of Autism and Developmental Disorders, 44(1), 120–136.  https://doi.org/10.1007/s10803-013-1856-1.CrossRefPubMedGoogle Scholar
  42. Ouellette-Kuntz, H. M., Coo, H., Lam, M., Yu, C. T., Breitenbach, M. M., Hennessey, P. E., et al. (2009). Age at diagnosis of autism spectrum disorders in four regions of Canada. Canadian Journal of Public Health, 100(4), 268–273.PubMedGoogle Scholar
  43. Ozonoff, S., Heung, K., Byrd, R., Hansen, R., & Hertz-Picciotto, I. (2008). The onset of autism: Patterns of symptom emergence in the first years of life. Autism Research, 1(6), 320–328.  https://doi.org/10.1002/aur.53.CrossRefPubMedPubMedCentralGoogle Scholar
  44. Ozonoff, S., Young, G. S., Carter, A., Messinger, D., Yirmiya, N., Zwaigenbaum, L., et al. (2011). Recurrence risk for autism spectrum disorders: A baby siblings research consortium study. Pediatrics.  https://doi.org/10.1542/peds.2010-2825.PubMedPubMedCentralGoogle Scholar
  45. Pandey, J., Verbalis, A., Robins, D. L., Boorstein, H., Klin, A., Babitz, T., et al. (2008). Screening for autism in older and younger toddlers with the Modified Checklist for Autism in Toddlers. Autism, 12(5), 513–535.  https://doi.org/10.1177/1362361308094503.CrossRefPubMedGoogle Scholar
  46. Penner, M. (2016). Policy analysis and evaluation of national clinician-reported practices for diagnosis of autism spectrum disorder. Toronto: University of Toronto.Google Scholar
  47. Perry, A., Cummings, A., Geier, J. D., Freeman, N. L., Hughes, S., Managhan, T., et al. (2011). Predictors of outcome for children receiving intensive behavioral intervention in a large, community-based program. Research in Autism Spectrum Disorders, 5(1), 592–603.  https://doi.org/10.1016/j.rasd.2010.07.003.CrossRefGoogle Scholar
  48. 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.CrossRefPubMedGoogle Scholar
  49. 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.  https://doi.org/10.1001/jamapediatrics.2016.2695.CrossRefPubMedGoogle Scholar
  50. Pierce, K., Carter, C., Weinfeld, M., Desmond, J., Hazin, R., Bjork, R., & Gallagher, N. (2011). Detecting, studying, and treating autism early: The one-year well-baby check-up approach. The Journal of Pediatrics, 159(3), 458–465.e6.  https://doi.org/10.1016/j.jpeds.2011.02.036.CrossRefPubMedPubMedCentralGoogle Scholar
  51. Pierce, K., Courchesne, E., & Bacon, E. (2016). To screen or not to screen universally for autism is not the question: Why the task force got it wrong. The Journal of Pediatrics, 176, 182–194.  https://doi.org/10.1016/j.jpeds.2016.06.004.CrossRefPubMedPubMedCentralGoogle Scholar
  52. Powell, C. M. (2016). Autism screening or smoke screen and mirrors? JAMA Neurology, 73(4), 386.  https://doi.org/10.1001/jamaneurol.2016.0126.CrossRefPubMedPubMedCentralGoogle Scholar
  53. Régie de l’assurance maladie du Québec. (2015). Manuel de facturation. rémunération à l’acte. Table B-Tarification des visites. Régle de l’assurance maladie Quebec. Retrieved April 25, 2016 from http://www.ramq.gouv.qc.ca/fr/professionnels/medecins-specialistes/manuels/Pages/facturation.aspx.
  54. Richards, C., Jones, C., Groves, L., Moss, J., & Oliver, C. (2015). Prevalence of autism spectrum disorder phenomenology in genetic disorders: A systematic review and meta-analysis. The Lancet Psychiatry, 2(10), 909–916.  https://doi.org/10.1016/S2215-0366(15)00376-4.CrossRefPubMedGoogle Scholar
  55. Robins, D., Adamson, L. B., Barton, M., Connell, J. E., Dumont-Mathieu, T., Dworkin, P. H., et al. (2016). Universal autism screening for toddlers: Recommendations at odds. Journal of Autism and Developmental Disorders, 46(5), 1880–1882.  https://doi.org/10.1007/s10803-016-2697-5.CrossRefPubMedPubMedCentralGoogle Scholar
  56. Robins, D., Fein, D., Barton, M. L., & Green, J. A. (2001). The Modified 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.CrossRefPubMedGoogle Scholar
  57. Sanders, G. D., Neumann, P. J., Basu, A., Brock, D. W., Feeny, D., Krahn, M., et al. (2016). Recommendations for conduct, methodological practices, and reporting of cost-effectiveness analyses: Second panel on cost-effectiveness in health and medicine. JAMA, 316(10), 1093.  https://doi.org/10.1001/jama.2016.12195.CrossRefPubMedGoogle Scholar
  58. Silverstein, M., & Radesky, J. (2016). Embrace the complexity: The US Preventive Services Task Force recommendation on screening for autism spectrum disorder. JAMA, 315(7), 661.  https://doi.org/10.1001/jama.2016.0051.CrossRefPubMedGoogle Scholar
  59. Siu, A. L., the US Preventive Services Task Force (USPSTF), Bibbins-Domingo, K., Grossman, D. C., Baumann, L. C., Davidson, K. W., et al. (2016). Screening for autism spectrum disorder in young children: US Preventive Services Task Force recommendation statement. JAMA, 315(7), 691.  https://doi.org/10.1001/jama.2016.0018.CrossRefPubMedGoogle Scholar
  60. Sparrow, S., Balla, D., & Cicchetti, D. (2005). Vineland adaptive behavior scales (2nd edn.). Bloomington, MN: Pearson Assessment.Google Scholar
  61. Statistics Canada. (2015a). Table 102–4505. Crude birth rate, age-specific and total fertility rates (live births), Canada, provinces and territories. Statistics Canada. Retrieved January 19, 2016 from http://www5.statcan.gc.ca/cansim/a26?lang=eng&id=1024505.
  62. Statistics Canada. (2015b). Table 282–0074. Labour force survey estimates (LFS), wages of employees by job permanence, union coverage, sex and age group. Retrieved January 19, 2016 from http://www5.statcan.gc.ca/cansim/a26?lang=eng&id=2820074.
  63. Veenstra-VanderWeele, J., & McGuire, K. (2016). Rigid, inflexible approach results in no recommendation for autism screening. JAMA Psychiatry, 73(4), 327.  https://doi.org/10.1001/jamapsychiatry.2016.0143.CrossRefPubMedGoogle Scholar
  64. Volkmar, F., Siegel, M., Woodbury-Smith, M., King, B., McCracken, J., & State, M. (2014). Practice parameter for the assessment and treatment of children and adolescents with autism spectrum disorder. Journal of the American Academy of Child & Adolescent Psychiatry, 53(2), 237–257.  https://doi.org/10.1016/j.jaac.2013.10.013.CrossRefGoogle Scholar
  65. Zwaigenbaum, L., Bauman, M. L., Choueiri, R., Kasari, C., Carter, A., Granpeesheh, D., et al. (2015). Early intervention for children with autism spectrum disorder nder 3 years of age: Recommendations for practice and research. Pediatrics, 136(Supplement), S60–S81.  https://doi.org/10.1542/peds.2014-3667E.CrossRefPubMedGoogle Scholar
  66. Zwaigenbaum, L., Bauman, M. L., Fein, D., Pierce, K., Buie, T., Davis, P. A., et al. (2015). Early screening of autism spectrum disorder: Recommendations for practice and research. Pediatrics, 136(Supplement), S41–S59.  https://doi.org/10.1542/peds.2014-3667D.CrossRefPubMedGoogle Scholar
  67. Zwaigenbaum, L., Bryson, S. E., Szatmari, P., Brian, J., Smith, I. M., Roberts, W., et al. (2012). Sex differences in children with autism spectrum disorder identified within a high-risk infant cohort. Journal of Autism and Developmental Disorders, 42(12), 2585–2596.  https://doi.org/10.1007/s10803-012-1515-y.CrossRefPubMedGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  • Tracy Yuen
    • 1
    • 2
  • Melissa T. Carter
    • 3
  • Peter Szatmari
    • 1
    • 2
    • 4
  • Wendy J. Ungar
    • 1
    • 2
  1. 1.Institute of Heath Policy, Management and EvaluationUniversity of TorontoTorontoCanada
  2. 2.Program of Child Health Evaluative SciencesThe Hospital for Sick Children Research InstituteTorontoCanada
  3. 3.Regional Genetics ProgramChildren’s Hospital of Eastern OntarioOttawaCanada
  4. 4.Centre for Addiction and Mental HealthTorontoCanada

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