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Cost-Effectiveness Analysis of Universal Screening for Biliary Atresia in Japan
Eri Hoshino1, Kensuke Moriwaki2, Kosuke Morimoto3
1Comprehensive Unit for Health Economic Evidence Review and Decision Support (CHEERS), Research Organization of Science and Technology, Ritsumeikan University, Kyoto, Japan; Graduate School of Public Health, St. Luke's International University, Kyoto, Japan.
Insights
Universal newborn screening for biliary atresia using stool color cards or direct bilirubin (DB) testing shows potential cost-effectiveness. Both screening methods improve liver transplant-free survival, with DB testing yielding more life-years gained.
Area of Science:
- Pediatric Gastroenterology
- Public Health Policy
- Health Economics
Background:
- Biliary atresia is a severe neonatal liver disease requiring early diagnosis and intervention.
- Current diagnostic approaches lack universal screening, leading to delayed treatment and poorer outcomes.
- Evaluating cost-effectiveness of screening strategies is crucial for resource allocation in healthcare.
Purpose of the Study:
- To assess the cost-effectiveness of universal newborn screening for biliary atresia in Japan.
- To compare stool color card and direct bilirubin (DB) testing against no screening.
- To determine the incremental cost-effectiveness ratio (ICER) for each strategy.
Main Methods:
- A decision analytic Markov microsimulation model was employed.
- Simulated outcomes for newborns under three strategies: stool color card, DB testing, and no screening.
- Analyzed event-free life-years (liver transplant-free survival), costs, and ICER over 25 years with a 2% discount rate.
Main Results:
- Stool color card screening increased event-free life-years by 44 compared to no screening, with an ICER of $339,258 per life-year gained.
- DB testing yielded 271 more event-free life-years than stool color card screening, at an ICER of $512,893 per life-year gained.
- DB testing resulted in 16 fewer liver transplants than stool color card screening, which had 2 fewer transplants than no screening.
Conclusions:
- Universal newborn screening for biliary atresia, particularly using DB testing, can be a cost-effective public health intervention.
- The cost-effectiveness is contingent upon the established willingness-to-pay thresholds for health benefits.
- Screening strategies significantly reduce the need for liver transplants, improving long-term patient outcomes.
Objective:
To evaluate the cost-effectiveness of universal newborn screening using stool color card or direct bilirubin (DB) testing when comparing with no screening for biliary atresia in Japanese setting.
Study Design:
A decision analytic Markov microsimulation model was developed to evaluate the universal screening for biliary atresia. Our screening strategies included stool color card, DB, or no screening. The outcomes of all newborns undergoing 3 strategies were simulated to analyze event-free life-years defined as liver transplant-free survival, costs, and incremental cost-effectiveness ratio (ICER) over a 25-year period with an annual discount rate of 2% applied for both costs and outcomes. A 1-way sensitivity analysis was performed to assess the uncertainty.
Results:
There were 941 000 newborn infants in our cohort and 114 cases of biliary atresia. The base case analysis showed that the stool color card strategy was $14 927 337 higher than no screening with an increase in 44 more event-free life-years gained, resulting in an ICER of $339 258 per event-free life-year gained. The DB screening strategy compared with stool color card was $138 994 060 higher with an increase in 271 more event-free life-years gained and an ICER of $512 893 per event-free life-year gained. The DB screening strategy compared with no screening resulted in an ICER of $488 639 per event-free life-year gained. The DB screening resulted in 16 fewer liver transplants than stool color card and stool color card had 2 fewer liver transplants than no screening.
Conclusions:
Universal screening for biliary atresia could be cost-effective depending on the willingness to pay thresholds for health benefits.
