Identifying the Optimum Strategy for Identifying Adults and Children With Celiac Disease: A Cost-Effectiveness and Value of Information Analysis

Keeney, Edna; Elwenspoek, Martha M.C.; Jackson, Joni; Roadevin, Cristina; Jones, Hayley E.; O’Donnell, Rachel; Sheppard, Athena L.; Dawson, Sarah; Lane, Deborah; Stubbs, Jo; Everitt, Hazel; Watson, Jessica C.; Hay, Alastair D.; Gillett, Peter; Robins, Gerry; Mallett, Sue; Whiting, Penny F.; Thom, Howard

Abstract

Objectives
Celiac disease (CD) is thought to affect around 1% of people in the United Kingdom, but only approximately 30% are diagnosed. The aim of this work was to assess the cost-effectiveness of strategies for identifying adults and children with CD in terms of who to test and which tests to use.

Methods
A decision tree and Markov model were used to describe testing strategies and model long-term consequences of CD. The analysis compared a selection of pre-test probabilities of CD above which patients should be screened, as well as the use of different serological tests, with or without genetic testing. Value of information analysis was used to prioritize parameters for future research.

Results
Using serological testing alone in adults, immunoglobulin A (IgA) tissue transglutaminase (tTG) at a 1% pre-test probability (equivalent to population screening) was most cost-effective. If combining serological testing with genetic testing, human leukocyte antigen combined with IgA tTG at a 5% pre-test probability was most cost-effective. In children, the most cost-effective strategy was a 10% pre-test probability with human leukocyte antigen plus IgA tTG. Value of information analysis highlighted the probability of late diagnosis of CD and the accuracy of serological tests as important parameters. The analysis also suggested prioritizing research in adult women over adult men or children.

Conclusions
For adults, these cost-effectiveness results suggest UK National Screening Committee Criteria for population-based screening for CD should be explored. Substantial uncertainty in the results indicate a high value in conducting further research.

Citation

Keeney, E., Elwenspoek, M. M., Jackson, J., Roadevin, C., Jones, H. E., O’Donnell, R., Sheppard, A. L., Dawson, S., Lane, D., Stubbs, J., Everitt, H., Watson, J. C., Hay, A. D., Gillett, P., Robins, G., Mallett, S., Whiting, P. F., & Thom, H. (2024). Identifying the Optimum Strategy for Identifying Adults and Children With Celiac Disease: A Cost-Effectiveness and Value of Information Analysis. Value in Health, 27(3), 301-312. https://doi.org/10.1016/j.jval.2023.12.010

• Journal Article Type: Article
• Acceptance Date: Dec 11, 2023
• Online Publication Date: Dec 26, 2025
• Publication Date: 2024-03
• Deposit Date: Feb 19, 2025
• Journal: Value in Health
• Print ISSN: 1098-3015
• Electronic ISSN: 1524-4733
• Publisher: Elsevier
• Peer Reviewed: Peer Reviewed
• Volume: 27
• Issue: 3
• Pages: 301-312
• DOI: https://doi.org/10.1016/j.jval.2023.12.010
• Public URL: https://nottingham-repository.worktribe.com/output/45593665
• Publisher URL: https://www.valueinhealthjournal.com/article/S1098-3015(23)06243-5/abstract?_returnURL=https%3A%2F%2Flinkinghub.elsevier.com%2Fretrieve%2Fpii%2FS1098301523062435%3Fshowall%3Dtrue
• Additional Information: This article is maintained by: Elsevier; Article Title: Identifying the Optimum Strategy for Identifying Adults and Children With Celiac Disease: A Cost-Effectiveness and Value of Information Analysis; Journal Title: Value in Health; CrossRef DOI link to publisher maintained version: https://doi.org/10.1016/j.jval.2023.12.010; Content Type: article; Copyright: © 2024 International Society for Pharmacoeconomics and Outcomes Research, Inc. Published by Elsevier Inc.