{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,19]],"date-time":"2026-09-19T00:21:26Z","timestamp":1789777286239,"version":"4.0.1"},"reference-count":45,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2024,5,23]],"date-time":"2024-05-23T00:00:00Z","timestamp":1716422400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2024,5,23]],"date-time":"2024-05-23T00:00:00Z","timestamp":1716422400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100009326","name":"Cancer Research Society","doi-asserted-by":"publisher","award":["23445"],"award-info":[{"award-number":["23445"]}],"id":[{"id":"10.13039\/100009326","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"published-print":{"date-parts":[[2024,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Li\u2013Fraumeni 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\u2010support tool provides a standardized approach to identify patients at risk of CPSs.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We conducted a cost\u2013utility analysis, from the healthcare payer perspective, to compare MIPOGG\u2010guided, physician\u2010guided, and universal genetic testing strategies to detect LFS in female patients diagnosed at an age of less than 18\u00a0years with osteosarcoma. We developed a decision tree and discrete\u2010event 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\u2010adjusted life\u2010years (QALYs), healthcare costs, and incremental cost\u2013utility ratios (ICURs). We conducted probabilistic and scenario analyses to assess the uncertainty surrounding model parameters.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Compared to the physician\u2010guided testing, the MIPOGG\u2010guided strategy was marginally more expensive by $105 (\u2212$516; $743), but slightly more effective by 0.003 (\u22120.04; 0.045) QALYs. Compared to MIPOGG, the universal testing strategy was $1333 ($732; $1953) more costly and associated with 0.011 (\u22120.043; 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.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>This study provides evidence for clinical and policy decision\u2010making on the cost\u2010effectiveness of genetic referral strategies to identify LFS in the setting of osteosarcoma. MIPOGG\u2010guided strategy was most likely to be cost\u2010effective at a willingness\u2010to\u2010pay threshold value of $50,000\/QALY.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/pbc.31077","type":"journal-article","created":{"date-parts":[[2024,5,23]],"date-time":"2024-05-23T23:44:57Z","timestamp":1716507897000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Cost\u2010effectiveness of the McGill interactive pediatric oncogenetic guidelines in identifying Li\u2013Fraumeni syndrome in female patients with osteosarcoma"],"prefix":"10.1002","volume":"71","author":[{"given":"Juan David","family":"Rios","sequence":"first","affiliation":[{"name":"Child Health Evaluative Sciences Peter Gilgan Centre for Research and Learning The Hospital for Sick Children  Toronto Ontario 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