Objective Biliary atresia, a rare newborn liver disease, is the most common cause of liver-related death in children and the main indication for paediatric liver transplantation. Early detection and surgical intervention with a Kasai portoenterostomy offers the best chance for long-term patient survival. We conducted a cost-effectiveness analysis to compare no universal screening with screening using either a home-based infant stool colour card with passive card distribution strategy, or conjugated bilirubin testing. Methods A Markov model was developed, with structure, costs, and probabilities informed by the literature and clinical expert opinion, to simulate a newborn cohort over a 10-year time horizon. Health benefits were expressed as life-years gained. This analysis was conducted from the perspective of the Canadian publicly funded health care system (all costs in Canadian dollars). Both deterministic and probabilistic analyses were conducted. Results Screening using a home-based colour card with passive card distribution was a cost-effective option. For a population of 392,902 annual births in Canada, this strategy cost approximately $192,000 more than no universal screening but led to eight life-years gained (incremental cost-effectiveness ratio (ICER) = $24,065 per life-year gained). Screening using conjugated bilirubin testing versus the colour card cost $2,369,199 more and led to five more life-years gained (ICER= $473,840 per life year gained), and so was not cost-effective. Conclusions A home-based screening program using infant stool colour cards with a passive distribution strategy could be highly cost-effective when administered at a low unit cost and with a reasonable screening performance.
Diagnostic Yield of Newborn Screening for Biliary Atresia Using Direct or Conjugated Bilirubin Measurements