OBJECTIVES:

Synovial sarcoma (SS) and myxoid/round cell liposarcoma (MRCLS) are rare subtypes of soft tissue sarcoma (STS). Outcomes for patients with advanced/metastatic SS/MRCLS (mSS/mMRCLS) are poor and systemic treatment options are limited. New cell therapies are being investigated for mSS/mMRCLS, and economic and utility evidence will be required by health technology assessment (HTA) bodies to evaluate these new treatments. The objective of this study was to systematically review the economic and utility evidence for previously treated mSS/mMRCLS.

METHODS:

Searches were conducted in Embase, MEDLINE, EconLit, Cochrane, and DARE (2000-2022), complemented with selected conferences (2020-2022) and HTA websites, published in English. Observational studies, clinical trials, and economic studies on patients with mSS/mMRCLS, advanced/metastatic STS (mSTS) results stratified by mSS/mMRCLS, or >80% study population with mSS/mMRCLS, were eligible for inclusion. mSTS population studies were flagged for additional consideration. No restriction was placed on interventions or comparators. Outcomes included economic evaluations, cost, healthcare resource use (HCRU), or utility values.

RESULTS:

Titles and abstracts of 1223 studies were screened, of these 92 full-text studies were reviewed. None of these studies met the inclusion criteria for the mSS/mMRCLS population; hence, no evidence was identified on the economic burden or utilities. However, for the broader mSTS population, 8 costs/HCRU studies, 3 utility studies, and 10 HTA assessments were identified. Cost drivers identified in literature included drug and inpatient hospitalization costs. HTA reviews noted evidence gaps for drug administration, wastage, adverse events, post-progression costs, and utilities for eribulin, pazopanib, olaratumab, and trabectedin.

CONCLUSIONS:

HCRU, cost, and utility evidence have focused on mSTS rather than on specific histology subtypes, resulting in limited data for mSS/mMRCLS. HTA reviews have also noted these evidence gaps in submissions for mSTS populations (that include mSS/mMRCLS). Further research is required to elucidate the impact of mSS/mMRCLS on patient utilities, and economic burden.