{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T04:59:04Z","timestamp":1785560344997,"version":"3.56.0"},"reference-count":90,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,7,31]],"date-time":"2026-07-31T00:00:00Z","timestamp":1785456000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,31]],"date-time":"2026-07-31T00:00:00Z","timestamp":1785456000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Intl Journal of Cancer"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Cancer predisposition syndromes (CPS), arising from germline pathogenic variants in cancer predisposition genes (CPGs), are increasingly recognized as major contributors to pediatric and adult malignancies. Recent genomic advances have significantly enhanced the diagnosis, surveillance, and management of CPS, providing a unique framework for cancer prevention\u00a0and interception of pre\u2010malignant lesions. Increased utilization of comprehensive germline and tumor\u2010normal sequencing\u00a0has improved diagnostic precision, allowing early identification of at\u2010risk individuals, including those with and without a family history of cancer. Integration of genome and long\u2010read sequencing technologies further increases diagnostic yield\u00a0by detecting structural and noncoding variants. Early diagnosis strategies, such as newborn screening, have demonstrated clinical utility and cost\u2010effectiveness in population studies, such as the\n                    <jats:italic>TP53<\/jats:italic>\n                    R337H variant newborn screening in Brazil. Evidence\u2010based surveillance protocols, supported by outcome data, have led to earlier tumor detection and improved survival. Novel \u2018liquid biopsy\u2019 approaches offer minimally invasive tools for surveillance, with the potential to detect malignancies prior to radiologic findings. Beyond early detection, emerging strategies aim to intercept tumor development. Medical prevention strategies such as metformin in Li\u2013Fraumeni syndrome and aspirin in Lynch syndrome show promise in modifying cancer risk. Immunoprevention, particularly neoantigen\u2010targeted vaccines in mismatch repair\u2010deficient tumors, is an evolving frontier with preliminary evidence of immunogenicity and safety. Together, these advances represent a paradigm shift in CPS management, offering a model for precision cancer prevention. Continued international collaboration, longitudinal studies, and health economic evaluations will be critical to translating these innovations into clinical and public health practice.\n                  <\/jats:p>","DOI":"10.1002\/ijc.70674","type":"journal-article","created":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T04:31:58Z","timestamp":1785558718000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Future Trends in Cancer Prevention and Interception in Cancer Predisposition Syndromes: Leveraging Insights From High\u2010Risk Populations"],"prefix":"10.1002","author":[{"given":"Yiming","family":"Wang","sequence":"first","affiliation":[{"name":"Division of Clinical and Metabolic Genetics The Hospital for Sick Children  Toronto Ontario Canada"},{"name":"Genetics and Genome Biology Program, The Hospital for Sick Children  Toronto Ontario Canada"},{"name":"Department of Pediatrics, Temerty Faculty of Medicine University of Toronto  Toronto Ontario Canada"},{"name":"Institute of Medical Science, Temerty Faculty of Medicine University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Malkin","sequence":"additional","affiliation":[{"name":"Genetics and Genome Biology Program, The Hospital for Sick Children  Toronto Ontario Canada"},{"name":"Department of Pediatrics, Temerty Faculty of Medicine University of Toronto  Toronto Ontario Canada"},{"name":"Institute of Medical Science, Temerty Faculty of Medicine University of Toronto  Toronto Ontario Canada"},{"name":"Department of Medical Biophysics, Temerty Faculty of Medicine University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0603-9139","authenticated-orcid":false,"given":"Steven M.","family":"Lipkin","sequence":"additional","affiliation":[{"name":"Joan and Sanford I. Weill Department of Medicine Weill Cornell Medical College  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,31]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"crossref","first-page":"820","DOI":"10.1073\/pnas.68.4.820","article-title":"Mutation and Cancer: Statistical Study of Retinoblastoma","volume":"68","author":"Knudson A. G.","year":"1971","journal-title":"Proceedings of the National Academy of Sciences of the United States of America"},{"key":"e_1_2_10_3_1","doi-asserted-by":"crossref","first-page":"e1","DOI":"10.1158\/1078-0432.CCR-17-0702","article-title":"Pediatric Cancer Predisposition and Surveillance: An Overview, and a Tribute to Alfred G. Knudson Jr","volume":"23","author":"Brodeur G. 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