A systematic literature review of the cost-effectiveness of treatments, costs, and resource use in patients with Burkitt lymphoma

Abstract

Background: Burkitt Lymphoma (BL) is a rare and aggressive type of non-Hodgkin lymphoma (NHL), accounting for 0.8% of all B-cell lymphomas, with five-year survival ranging from 48% to 87%. Currently, intensive short-cycle and low-intensity multiagent immunochemotherapy regimens are used, however, it is not clear what health economic evidence exists for treatments in BL. Thereby, a systematic literature review (SLR) was conducted to understand the economic (costs and resource-use, economic evaluations) evidence base for patients with BL. Methods: AnSLR was conducted using EmbaseVR, PubMedVR, and EconLit from database inception through June 2019. Results: A total of 758 abstracts and 132 full-texts were screened. We identified 2 economic evaluations and 7 studies reporting data for costs or resource-use covering four countries (Malawi, Nigeria, Uganda, Netherlands). The economic evidence varied in different regions due to unbalanced socio-economic development. The median length of hospital stay ranged from 13 days in Nigeria to 134 days in the Netherlands. The patients were also reluctant to healthcare and took 4-8 weeks from the onset of symptoms to present at the treatment centers in Nigeria. The total direct cost of treating BL was US$103.8 (Nigeria), US$350.14 (Uganda), and US$7342.17 (Netherlands) per patient. Dominant drivers for cost were the length of in-hospital stay and medication costs. Both the economic evaluations presented models for paediatric patients with no treatment as the comparator of choice and a continuous discount rate of 3%. Models were built from a governmental perspective with discounting and duration of treatment significantly impacting the sensitivity analysis. The cost per DALY averted was US$97 in Uganda and US$14,243 in Malawi, reflecting very cost-effective chemothera-peutic interventions. Conclusions: This review highlights the limited literature on the economic dimensions of BL treatment, however, also demonstrates cost-effective interventions in resource-limited settings. There remains a need for future research and careful consideration of BL as an investment in health systems strengthening and improving the health outcomes.