Burden of Myelodysplastic Syndromes Part II: Systematic Literature Review of Economic Burden

BACKGROUND: Myelodysplastic syndromes (MDS) are malignant clonal neoplastic bone marrow disorders characterized by ineffective hematopoiesis, leading to abnormal and/or dysfunctional blood cells that express as different forms of cytopenia. A comprehensive assessment of the disease burden is lacking.

OBJECTIVES: This review aimed to systematically identify, review, and synthesize the evidence for healthcare resource utilization and costs associated with MDS as part of assessing the burden of the disease.

METHODS: A systematic literature search was implemented using MEDLINE and EMBASE from inception to July 13, 2021, to identify eligible studies that reported costs associated with MDS. Two reviewers screened and reviewed all articles independently and in duplicate. All costs are presented as annual cost, and were converted to 2020 US$ using inflation and exchange rates. Considering the heterogeneity in treatments, disease risk, and study design, a descriptive analysis was conducted.

RESULTS: A total of 7539 records were identified in the initial search. After completing title/abstract and full-text screenings, 49 unique studies from 13 countries were included in the review: 1 randomized controlled trial (sample size n=60), 23 observational studies (n=24 to 9524), and 25 database analyses (n=31 to 885,726). The time frame of studies ranged from 1992 to 2018. Overall, the total annual cost of MDS ranged from $16,821 to $243,802. The median (range) cost for higher-risk MDS was $172,707 ($47,260 to $227,758) vs $163,285 ($21,502 to $223,714) for lower-risk MDS. For patients treated with hypomethylating agents, the median (range) total cost was $160,523 ($33,506 to $243,802) vs $56,857 ($16,821 to $233,714) for those treated with other regimens. The length of stay in hospital varied from 1.6 to 52.8 days, with the hospitalization cost ranging from $6019 to $32,526.

CONCLUSIONS: The economic burden of MDS was substantial regardless of the risk profile. The costs varied substantially across treatment regimens.