OBJECTIVES: Osteosarcoma is the most common bone tumour that arises from mesenchymal cells and characterised by areas of uncontrolled or abnormal bone growth. Chemotherapy and surgery have been a major treatment option for osteosarcoma. The objective of this systematic review was to identify and summarise model-based economic evaluations of treatments in patients with osteosarcoma.

METHODS: Embase.com was searched in accordance with PRISMA guidelines to identify English language studies published from 2013 to 2024, reporting economic evaluations in patients with osteosarcoma. Electronic searches were supplemented by bibliographic searches; publications were screened by two independent reviewers.

RESULTS: A total of 810 potentially relevant records were screened, five met the inclusion criteria, all evaluating mifamurtide. Two studies were conducted in Korea, while one study each was conducted in the UK, Spain and Mexico. All studies utilised a Markov model for their analyses. One study each adopted the perspectives of the Spanish National Health Service, Mexican public health institutions and the UK National Health Service; while it was not reported in two studies. In Mexico and Spain, mifamurtide plus chemotherapy was a cost-effective option compared with chemotherapy alone with an ICER of $55,837/QALY and €76,620/QALY, respectively. In the UK, ICER for maintenance chemotherapy plus mifamurtide over maintenance chemotherapy alone was £68,734/QALY. In Korea, an increase of 45.2% and 10.5% was observed in QALY with the addition of mifamurtide to chemotherapy in patients with and without metastatic osteosarcoma, respectively. Another Korean study reported that, mifamurtide plus chemotherapy versus chemotherapy alone resulted in incremental QALYs of 1.57.

CONCLUSIONS: The literature search identified only one intervention, mifamurtide, with economic evaluations in osteosarcoma and thus, indicating a significant unmet need. Mifamurtide in combination with chemotherapy was a cost-effective option compared to chemotherapy alone. Further research is essential to assess the economic impact of emerging therapies for this rare disease.