{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T16:29:54Z","timestamp":1793723394556,"version":"4.6.1"},"reference-count":102,"publisher":"The Journal of Health Economics and Outcomes Research","issue":"2","license":[{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["jheor.org"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:sec id=\"background\">\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hereditary cancer syndromes account for approximately 10% of all cancers. Many of these, classified as rare tumor risk syndromes (RTRS), lack comprehensive data on natural history, lifetime cancer risk, and the long-term impact of surveillance. These evidence gaps limit the development of decision-analytic disease progression models and cost-effectiveness analyses needed to support healthcare decision-making.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"objective\">\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To map the available evidence on the natural history, cancer risk, and surveillance strategies in selected RTRS to inform disease progression models and future economic evaluations.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"methods\">\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A scoping review was conducted on 8 RTRS: Birt-Hogg-Dub\u00e9 syndrome, familial gastrointestinal stromal tumor, familial melanoma, hereditary diffuse gastric cancer, hereditary leiomyomatosis and renal cell cancer, Li-Fraumeni syndrome, Peutz-Jeghers syndrome, and PTEN hamartoma tumor syndrome. Systematic searches of PubMed, Embase, and relevant gray literature sources were performed between April and June 2026 for studies published since 2010.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"results\">\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>After screening 16\u2009631 records, 80 studies were included in the review. Findings indicate significant heterogeneity in data availability and surveillance effectiveness. Some RTRS have relatively well-defined surveillance recommendations, whereas others remain poorly characterized. Across studies, cancer incidence was markedly higher and occurred earlier than in the general population, with lifetime cancer risks approaching 100% in certain syndromes. However, only 44% of studies reported follow-up protocols and fewer than 12% compared surveillance with expectant management. Limited data on postdiagnosis survival further complicates economic evaluations of alternative care strategies.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"conclusions\">\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The evidence base for RTRS is heterogeneous, particularly regarding longitudinal outcomes and comparative surveillance strategies. These limitations restrict the parameterization of disease progression models and the evaluation of the cost-effectiveness of surveillance strategies, leaving substantial uncertainty about optimal management strategies. Significant evidence gaps remain in the natural history and management of RTRS. This review provides a foundation for future cost-effectiveness analyses by mapping existing evidence and identifying critical knowledge gaps in RTRS care.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.36469\/001c.167564","type":"journal-article","created":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T14:55:41Z","timestamp":1791816941000},"update-policy":"https:\/\/doi.org\/10.36469\/00001","source":"Crossref","is-referenced-by-count":0,"title":["Natural History of Rare Tumor Risk Syndromes: A Scoping Review to Inform Cost-Effectiveness Models"],"prefix":"10.36469","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2313-475X","authenticated-orcid":true,"given":"Carolina","family":"Di Fabrizio","sequence":"first","affiliation":[{"name":"NOVA School of Business and Economics, NOVA University of Lisbon, Carcavelos, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judite","family":"Gon\u00e7alves","sequence":"additional","affiliation":[{"name":"NOVA School of Business and Economics, NOVA University of Lisbon, Carcavelos, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zillur R.","family":"Shabuz","sequence":"additional","affiliation":[{"name":"Lancaster School of Mathematical Sciences, Lancaster University, Bailrigg, Lancaster, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"Titman","sequence":"additional","affiliation":[{"name":"School of Mathematical Sciences, Lancaster University, Bailrigg, Lancaster, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paula","family":"Boaventura","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/04wjk1035","id-type":"ROR","asserted-by":"publisher"}],"name":"i3S \u2013 Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Svetlana","family":"Bajalica Lagercrantz","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/056d84691","id-type":"ROR","asserted-by":"publisher"}],"name":"Karolinska Institutet, Department of Oncology-Pathology, Stockholm, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Peleteiro","sequence":"additional","affiliation":[{"name":"Hospital Epidemiology Center, Unidade Local de Sa\u00fade S\u00e3o Jo\u00e3o, Porto, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara","family":"Pereira","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/04wjk1035","id-type":"ROR","asserted-by":"publisher"}],"name":"i3S \u2013 Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liliana","family":"Sousa","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/04wjk1035","id-type":"ROR","asserted-by":"publisher"}],"name":"i3S \u2013 Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal"},{"id":[{"id":"https:\/\/ror.org\/03pbpa834","id-type":"ROR","asserted-by":"publisher"}],"name":"ERN GENTURIS - 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