Cost-Effectiveness Analysis of Large-Scale Screening for Celiac Disease in the Pediatric Population

OBJECTIVES: Celiac disease (CD) affects 1-3% of the US population. A negative HLA genetic test can rule out CD, but a positive test is non-specific. CD can be symptomatic or asymptomatic, making clinical detection challenging without mass screening. Our objective was to estimate the cost and cost-effectiveness of different CD screening strategies for mass screening in children.

METHODS: A healthcare system perspective was taken, which included direct medical costs. Average paid claims were used as a proxy for the cost of serologic and diagnostic tests. Three screening options for CD were explored through decision tree modeling with different combinations of genetic and serial serologic screening with tissue transglutaminase (tTGA). We combined performance metrics for screening methods with real-world evidence through the Autoimmunity Screening for Kids study. Decision tree modeling was bridged to a two-state Markov model to evaluate the cost-per-case-detected and the incremental cost-effectiveness ratio (ICER) with quality-adjusted-life-years (QALY) as the health benefit in 2023 US dollars (USD). Future costs and outcomes were discounted at 3% per year. Sensitivity analyses were also performed.

RESULTS: Genetic screening with subsequent tTGA testing for those genetically at-risk cost-per-case detected was $2449 USD. tTGA testing up to three times in childhood cost-per-case detected was $1880 USD.

The ICER for CD screening was $1546 USD per QALY gained. Key influential inputs such as health utility score of treated CD had the largest impact on outcomes. The model was also sensitive to costs per each arm (screening vs no screening).

CONCLUSIONS: Serial testing with tTGA is less expensive than initial genetic screening followed by tTGA testing for general population CD screening, and likely cost-effective using an ICER threshold of $100,000 USD per QALY. CD mass screening in children increases costs to the health system but improves health benefits by improving quality of life for patients with CD.