دورية أكاديمية
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Cost-effectiveness of the McGill interactive pediatric oncogenetic guidelines in identifying Li-Fraumeni syndrome in female patients with osteosarcoma.

التفاصيل البيبلوغرافية
العنوان: Cost-effectiveness of the McGill interactive pediatric oncogenetic guidelines in identifying Li-Fraumeni syndrome in female patients with osteosarcoma.
المؤلفون: Rios JD; Child Health Evaluative Sciences, Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, Toronto, Ontario, Canada., Simbulan F; Child Health Evaluative Sciences, Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, Toronto, Ontario, Canada.; Institute of Health Policy, Management and Evaluation, Dalla Lana School of Public Health, University of Toronto, Toronto, Ontario, Canada., Reichman L; Child Health and Human Development, Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada., Caswell K; Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada., Tachdjian M; Child Health and Human Development, Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada., Malkin D; Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada.; Division of Haematology/Oncology, The Hospital for Sick Children, Toronto, Ontario, Canada.; Genetics and Genome Biology Program, The Hospital for Sick Children, Toronto, Ontario, Canada., Cotton C; Department of Statistics and Actuarial Sciences, University of Waterloo, Waterloo, Ontario, Canada., Nathan PC; Child Health Evaluative Sciences, Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, Toronto, Ontario, Canada.; Institute of Health Policy, Management and Evaluation, Dalla Lana School of Public Health, University of Toronto, Toronto, Ontario, Canada.; Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada.; Division of Haematology/Oncology, The Hospital for Sick Children, Toronto, Ontario, Canada., Goudie C; Child Health and Human Development, Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada.; Department of Pediatrics, Division of Hematology-Oncology, McGill University Health Centre, Montreal, Quebec, Canada., Pechlivanoglou P; Child Health Evaluative Sciences, Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, Toronto, Ontario, Canada.; Institute of Health Policy, Management and Evaluation, Dalla Lana School of Public Health, University of Toronto, Toronto, Ontario, Canada.
المصدر: Pediatric blood & cancer [Pediatr Blood Cancer] 2024 Aug; Vol. 71 (8), pp. e31077. Date of Electronic Publication: 2024 May 23.
نوع المنشور: Journal Article
اللغة: English
بيانات الدورية: Publisher: John Wiley Country of Publication: United States NLM ID: 101186624 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1545-5017 (Electronic) Linking ISSN: 15455009 NLM ISO Abbreviation: Pediatr Blood Cancer Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Hoboken, N.J. : John Wiley, c 2004-
مواضيع طبية MeSH: Osteosarcoma*/economics , Osteosarcoma*/genetics , Osteosarcoma*/diagnosis , Li-Fraumeni Syndrome*/genetics , Li-Fraumeni Syndrome*/diagnosis , Li-Fraumeni Syndrome*/economics , Cost-Benefit Analysis*, Humans ; Female ; Child ; Adolescent ; Bone Neoplasms/genetics ; Bone Neoplasms/economics ; Genetic Testing/economics ; Genetic Testing/methods ; Practice Guidelines as Topic ; Quality-Adjusted Life Years ; Breast Neoplasms/genetics ; Breast Neoplasms/economics
مستخلص: Background: Li-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.
Methods: We 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.
Results: Compared 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.
Discussion: This 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.
(© 2024 The Authors. Pediatric Blood & Cancer published by Wiley Periodicals LLC.)
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معلومات مُعتمدة: 23445 Cancer Research Society; FDN143234 Canadian Institutes for Health Research
فهرسة مساهمة: Keywords: Li–Fraumeni syndrome; MIPOGG; cost–utility analysis; osteosarcoma
تواريخ الأحداث: Date Created: 20240524 Date Completed: 20240626 Latest Revision: 20240626
رمز التحديث: 20240627
DOI: 10.1002/pbc.31077
PMID: 38783403
قاعدة البيانات: MEDLINE
الوصف
تدمد:1545-5017
DOI:10.1002/pbc.31077