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Review
. 2022 Oct 18:14:665-682.
doi: 10.2147/CEOR.S370311. eCollection 2022.

The Cost of Neurodevelopmental Disability: Scoping Review of Economic Evaluation Methods

Sanjeewa Kularatna  1 , Amarzaya Jadambaa  1 , Sameera Senanayake  1 , David Brain  1 , Nadia Hawker  2 , Nadine A Kasparian  3   4 , Bridget Abell  1 , Benjamin Auld  5 , Karen Eagleson  5 , Robert Justo  5 , Steven M McPhail  1   6
Affiliations
Review

The Cost of Neurodevelopmental Disability: Scoping Review of Economic Evaluation Methods

Sanjeewa Kularatna et al. Clinicoecon Outcomes Res. .

Abstract

The provision of effective care models for children with neurodevelopmental delay or disability can be challenging in resource constrained healthcare systems. Economic evaluations have an important role in informing resource allocation decisions. This review systematically examined the scope and methods of economic models evaluating interventions for supporting neurodevelopment among children with common neurodevelopmental disorders and identified methods of economic models and presented policy implications. This scoping review employed the Arksey and O'Malley framework and aligned with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR). Four electronic databases were systematically searched to identify eligible model-based economic evaluations of neurodevelopmental care models published since 2000. The Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist was used to assess quality of reporting. Data were systematically extracted, tabulated, and qualitatively synthesised across diagnostic categories. Searches identified 1431 unique articles. Twelve studies used a decision analytic model to evaluate care for neurodevelopmental disorders and were included in the review. Included studies focused on attention-deficit/hyperactivity disorder (ADHD, n=6), autism spectrum disorder (ASD, n=3), cerebral palsy (n=2), and dyslexia (n=1). The most used decision analytic modelling approach was a Markov model (n=6), followed by a decision tree (n=3), and a combination of decision tree and Markov model (n=3). Most studies (n=7) adopted a societal perspective for reporting costs. None of the reviewed studies modelled impact on families and caregivers. Four studies reported cost-savings, three identified greater quality of life, and three identified cost increases.

Keywords: attention-deficit/hyperactivity disorder; decision analytic models; economic evaluation; neurodevelopmental disorders.

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Conflict of interest statement

Dr Bridget Abell reports grants from MRFF (Australian Government Competitive Grant), during the conduct of the study. The authors report no other conflicts of interest in this work.

Figures

Figure 1
Figure 1
PRISMA flow diagram showing the process of study selection for review inclusion. Adapted from Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. Creative Commons.
See this image and copyright information in PMC

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Grants and funding

This study is part of a wider program of research which is funded by a competitive grant via the National Health and Medical Research Council’s Medical Research Future Fund (ARGCHDGO0035). The funder had no role in the design of the study and data collection, analysis and interpretation of results and in writing the manuscript or submitting for publication.

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