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Screening for Celiac Disease in Childhood: Cost-Effectiveness of Multiple Genetic and Serological Testing Approaches
Jani Mäkinen1, Paula Heikkilä2, Juha Pajula3
1Celiac Disease Research Center, Tampere University, Tampere, Finland; Tampere Center for Child, Adolescent and Maternal Health Research, Tampere University, Tampere, Finland.
Insights
The most cost-effective strategy for childhood celiac disease (CD) screening is a single, untargeted serological test at age 11. This approach balances effectiveness and cost, though regional factors may influence optimal implementation.
Area of Science:
- Pediatric Gastroenterology
- Public Health
- Health Economics
Background:
- Celiac disease (CD) is significantly underdiagnosed, leading to prolonged diagnostic delays for affected children.
- Population-based screening is proposed to improve early detection, but cost-effectiveness remains a key consideration.
Purpose of the Study:
- To determine the most cost-effective screening strategy for childhood celiac disease.
- To evaluate various screening scenarios, including age, frequency, and genetic testing integration.
Main Methods:
- A Markov model utilizing Swedish data compared 272 screening strategies against no screening.
- Evaluated single-time vs. repeated serological testing (ages 3-18) and preliminary genetic testing.
- Sensitivity analysis assessed factors influencing optimal strategy and willingness-to-pay thresholds (€20,000/QALY).
Main Results:
- Single-time, untargeted screening at age 11 was the most cost-effective strategy in the default scenario.
- Repeated screening at ages 4 and 11 was also effective, particularly if CD prevalence exceeded 5% or screening costs were halved.
- Diagnostic delay was identified as the primary driver for optimal screening age.
Conclusions:
- Single-time, untargeted serological screening at age 11 is the recommended cost-effective strategy for childhood celiac disease.
- Optimal screening approaches may vary based on regional prevalence and economic factors.
- Repeated screening may be beneficial for high-risk populations or with higher willingness-to-pay thresholds.
Background & Aims:
Celiac disease is severely underdiagnosed, and many identified patients suffer from a long diagnostic delay. Population-based screening could improve this under-recognition, but its practical implementation remains unclear. We studied the most cost-effective screening strategy for celiac disease in childhood.
Methods:
A Markov model using primarily Swedish data was developed for comparing the cost-effectiveness of various scenarios. Key considerations included serological testing across ages 3 to 18 years, single-time vs repeated screening, and preliminary genetic testing to identify the at-risk population. Outcomes of the resulting 272 strategies were compared against a no-screening alternative using the incremental cost-effectiveness ratio. Sensitivity analysis was conducted at a €20,000 willingness to pay threshold to determine key factors influencing the optimal screening age(s) and conditions where repeated screening would be preferred.
Results:
Untargeted single-time screening was more cost-effective than repeated screening or the combined use of genetic testing in the default scenario. Single-time screening was most cost-effective at age 11 (95% confidence interval, 10-14 years), and repeated screening at ages 4 and 11. Repeated screening was preferred if prevalence of celiac disease at age 12 was ≥5%, diagnosis improved quality of life for asymptomatic patients, screening costs were halved, or a willingness to pay of €50,000 per quality-adjusted life year was adopted. Diagnostic delay was the key factor influencing the optimal screening age(s).
Conclusions:
The most cost-effective screening strategy for celiac disease was single-time untargeted serological testing at age 11. The optimal approach is affected by regional factors and may evolve with new evidence. Repeated screening may be preferable for high-risk groups and with high willingness to pay.
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