Presentation (CTI)

OBJECTIVES: Cholangiocarcinoma (CCA) is a biliary tract malignancy, with over 200,000 new cases estimated globally each year. Incidence is increasing worldwide, particularly in east and southeast Asia. Diagnosis is often delayed, and treatment pathways are complex with limited curative options, contributing to a high disease burden and significant healthcare costs. Additionally, economic evidence supporting their use remains scarce. This review aims to map health economic evaluations (EEs) of prevention and management strategies for CCA.
METHODS: Eligible studies included EEs related to CCA prevention or management. Non-English language studies were excluded. A systematic search of MEDLINE, Embase, EconLit, iHTA, and Tufts CEA was conducted from inception to May 2025. Study quality was assessed using CHEERS checklist. Two reviewers independently performed study selection, data extraction, and quality assessment following PRISMA guidelines. The strategies were categorized based on management pathway. Methodological characteristics, were narratively synthesised.
RESULTS: Overall, 21 studies met the eligibility criteria. One-third (n=7) conducted in the United States, followed by China (n=4), Taiwan (n=4), Thailand (n=3), Japan (n=2) and the United Kingdom (n=1). Evaluated strategies included surveillance and diagnostic strategies (n=3), curative treatments (n=2), chemotherapy and locoregional treatments (n=12), and palliative care (n=4). Methodological heterogeneity was common across studies, particularly perspectives and time horizons. Partitioned survival model was the most frequently used modelling type (42%), with two studies conducted economic evaluation alongside clinical trials. None of the studies evaluated the entire disease pathway. Variation on disease staging influenced economic outcomes. Stakeholder engagement and heterogeneity analysis were inconsistently reported.
CONCLUSIONS: There is increasing global interest in the EEs of CCA strategies. While a range of strategies have been assessed, none have evaluated the full disease pathway. This limits the ability to fully capture the complexity of CCA care. Comprehensive and methodologically robust evaluations are needed to inform optimal resource allocation for this high-burden but rare cancer.