Cost‐effectiveness of the McGill interactive pediatric oncogenetic guidelines in identifying Li–Fraumeni syndrome in female patients with osteosarcoma

Abstract

AbstractBackgroundLi–Fraumeni syndrome (LFS) is a penetrant cancer predisposition syndrome (CPS) associated with the development of many tumor types in young people including osteosarcoma and breast cancer (BC). The McGill Interactive Pediatric OncoGenetic Guidelines (MIPOGG) decision‐support tool provides a standardized approach to identify patients at risk of CPSs.MethodsWe conducted a cost–utility analysis, from the healthcare payer perspective, to compare MIPOGG‐guided, physician‐guided, and universal genetic testing strategies to detect LFS in female patients diagnosed at an age of less than 18 years with osteosarcoma. We developed a decision tree and discrete‐event simulation model to simulate the clinical and cost outcomes of the three genetic referral strategies on a cohort of female children diagnosed with osteosarcoma, especially focused on BC as subsequent cancer. Outcomes included BC incidence, quality‐adjusted life‐years (QALYs), healthcare costs, and incremental cost–utility ratios (ICURs). We conducted probabilistic and scenario analyses to assess the uncertainty surrounding model parameters.ResultsCompared to the physician‐guided testing, the MIPOGG‐guided strategy was marginally more expensive by $105 (−$516; $743), but slightly more effective by 0.003 (−0.04; 0.045) QALYs. Compared to MIPOGG, the universal testing strategy was $1333 ($732; $1953) more costly and associated with 0.011 (−0.043; 0.064) additional QALYs. The ICUR for the MIPOGG strategy was $33,947/QALY when compared to the physician strategy; the ICUR for universal testing strategy was $118,631/QALY when compared to the MIPOGG strategy.DiscussionThis study provides evidence for clinical and policy decision‐making on the cost‐effectiveness of genetic referral strategies to identify LFS in the setting of osteosarcoma. MIPOGG‐guided strategy was most likely to be cost‐effective at a willingness‐to‐pay threshold value of $50,000/QALY.