OBJECTIVES: Biliary tract cancer (BTC) encompasses a group of rare and aggressive malignancies, including cholangiocarcinoma and gallbladder cancer. This systematic literature review (SLR) investigates the economic burden of advanced/metastatic BTC on healthcare systems in major developed markets.

METHODS: Embase®, MEDLINE®, MEDLINE® In-Process, EconLit®, INAHTA, and the Centre for Reviews and Dissemination York were searched on 12-April-2024, for literature published since 2013. Documents underwent screening following Cochrane guidance by two independent reviewers, with conflicts resolved by a third, independent reviewer. Inclusion criteria: adult patients with advanced/metastatic BTC, and studies reporting direct healthcare costs (including treatment costs) and/or resource utilization, including economic evaluations. All results were converted and inflated to 2024 USD.

RESULTS: Of 1,728 citations retrieved, 13 studies, mostly from the US and Japan, met the inclusion criteria. Most studies reported on BTC with 5 studies focussed on cholangiocarcinoma. The included studies report heterogeneous outcomes. A retrospective US claims analysis found that the mean cholangiocarcinoma-related total direct monthly healthcare costs in patients was $9,477. A separate retrospective cost and resource use analysis using US private payer Cancer Care Quality Program data reported an all-cause per member per month healthcare cost for first-line patients of $23,124. Cost-effectiveness studies reported monthly costs per patient from US and China ranging from $3,568 - $9,876. Another study using the National Inpatient Sample reported mean monthly hospital charges of $10,564 for cholangiocarcinoma admissions. In Japan, a retrospective claims data analysis estimated the mean monthly all-cause healthcare costs of patients with metastatic BTC to be $4,118, while secondary studies reported monthly costs per patient of $12,818.

CONCLUSIONS: The economic burden of BTC varied widely between studies with monthly disease-related direct cost ranging from $3,500 - $23,000 in most countries, representing a significant burden to healthcare systems.