{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T02:34:34Z","timestamp":1791858874233,"version":"4.3.4"},"reference-count":34,"publisher":"Wiley","issue":"15","license":[{"start":{"date-parts":[[2022,6,1]],"date-time":"2022-06-01T00:00:00Z","timestamp":1654041600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100013956","name":"Aard- en Levenswetenschappen, Nederlandse Organisatie voor Wetenschappelijk Onderzoek","doi-asserted-by":"publisher","award":["016.196.338"],"award-info":[{"award-number":["016.196.338"]}],"id":[{"id":"10.13039\/501100013956","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003246","name":"Nederlandse Organisatie voor Wetenschappelijk Onderzoek","doi-asserted-by":"publisher","award":["016.196.338"],"award-info":[{"award-number":["016.196.338"]}],"id":[{"id":"10.13039\/501100003246","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005622","name":"Health Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005622","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2022,8]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Women with <jats:italic>PTEN<\/jats:italic> Hamartoma Tumor Syndrome (PHTS) are offered breast cancer (BC) surveillance because of an increased BC lifetime risk. Surveillance guidelines are, however, expert opinion\u2013based because of a lack of data. We aimed to assess the yield and effectiveness of BC surveillance and the prevalence and type of breast disease in women with PHTS.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Sixty\u2010five women with PHTS who visited our center between 2001 and 2021 were included. Surveillance consisted of annual magnetic resonance imaging (MRI) and mammography from ages 25 and 30\u2009years, respectively.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Thirty\u2010nine women enrolled in the BC surveillance program (median age at first examination, 38\u2009years [range, 24\u201370]) and underwent 156 surveillance rounds. Surveillance led to detection of BC in 7\/39 women (cancer detection rate [CDR], 45\/1000 rounds) and benign breast lesions (BBLs) in 11\/39 women. Overall sensitivity<jats:sub>2<\/jats:sub> (which excludes prophylactic\u2010mastectomy detected BCs) was 100%, whereas sensitivity<jats:sub>2<\/jats:sub> of mammography and MRI alone was 50% and 100%, respectively. Overall specificity was higher in follow\u2010up rounds (86%) versus first rounds (71%). Regardless of surveillance, 21\/65 women developed 35 distinct BCs (median age at first diagnosis, 40\u2009years [range, 24\u201359]) and 23\/65 developed 89 BBLs (median age at first diagnosis, 38\u2009years [range, 15\u201361]). Surveillance\u2010detected BCs were all T1 and N0, whereas outside surveillance\u2010detected BCs were more often \u2265T2 (60%) and N+ (45%) (<jats:italic>p<\/jats:italic>\u2009&lt;\u2009.005).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The findings show that annual BC surveillance with MRI starting at age 25\u2009years enables detection of early\u2010stage BCs. Performance measures of surveillance and CDR were both high. BBLs were commonly present, underlining the importance of evaluation of all lesions independently.<\/jats:p><\/jats:sec><jats:sec><jats:title>Lay summary<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Breast cancer surveillance leads to decreased tumor stage and improved survival.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Breast cancer surveillance with breast magnetic resonance imaging from age 25\u2009years onward is recommended.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1002\/cncr.34326","type":"journal-article","created":{"date-parts":[[2022,6,1]],"date-time":"2022-06-01T13:18:32Z","timestamp":1654089512000},"page":"2883-2891","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":16,"title":["The yield and effectiveness of breast cancer surveillance in women with <scp>PTEN<\/scp> Hamartoma Tumor Syndrome"],"prefix":"10.1002","volume":"128","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8553-863X","authenticated-orcid":false,"given":"Alma","family":"Hoxhaj","sequence":"first","affiliation":[{"name":"Department of Imaging Radboud University Medical Center  Nijmegen The Netherlands"},{"name":"Department of Radiology and Nuclear Medicine the Netherlands Cancer Institute, Antoni van Leeuwenhoek Hospital  Amsterdam The Netherlands"},{"name":"Radboud University Medical Center Radboud Institute for Health Sciences  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meggie M.C.M.","family":"Drissen","sequence":"additional","affiliation":[{"name":"Radboud University Medical Center Radboud Institute for Health Sciences  Nijmegen The Netherlands"},{"name":"Department of Human Genetics Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janet R.","family":"Vos","sequence":"additional","affiliation":[{"name":"Radboud University Medical Center Radboud Institute for Health Sciences  Nijmegen The Netherlands"},{"name":"Department of Human Genetics Radboud University Medical Center  Nijmegen The Netherlands"},{"name":"European Reference Network Genetic Tumour Risk Syndromes (ERN GENTURIS)  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Bult","sequence":"additional","affiliation":[{"name":"Department of Pathology Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ritse M.","family":"Mann","sequence":"additional","affiliation":[{"name":"Department of Imaging Radboud University Medical Center  Nijmegen The Netherlands"},{"name":"Department of Radiology and Nuclear Medicine the Netherlands Cancer Institute, Antoni van Leeuwenhoek Hospital  Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicoline","family":"Hoogerbrugge","sequence":"additional","affiliation":[{"name":"Radboud University Medical Center Radboud Institute for Health Sciences  Nijmegen The Netherlands"},{"name":"Department of Human Genetics Radboud University Medical Center  Nijmegen The Netherlands"},{"name":"European Reference Network Genetic Tumour Risk Syndromes (ERN GENTURIS)  Nijmegen The Netherlands"},{"name":"Department of Pathology Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,6]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnci\/djt277"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/humu.23636"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers11060844"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-013-9674-3"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2012-101339"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.Ccr-11-2283"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13875"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10897-008-9187-7"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-med-052218-125823"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.3322\/canjclin.57.2.75"},{"key":"e_1_2_9_12_1","volume-title":"Guideline PTEN hamartoma tumour syndrome. Version 1.0","author":"Dutch Society of Clinical Genetics (VKGN)","year":"2015"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10549-020-05642-1"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1086\/301749"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13058-018-1019-6"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41431-020-0651-7"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ejhg.5200289"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1200\/jco.2014.59.0463"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-018-0110-6"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00330-008-0863-7"},{"key":"e_1_2_9_21_1","volume-title":"ACR BI\u2010RADS Atlas, Breast Imaging Reporting and Data System","author":"D'Orsi CJSE","year":"2013"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/(sici)1097\u20100258(19980430)17:8<857::aid\u2010sim777>3.0.co;2\u2010e"},{"key":"e_1_2_9_23_1","volume-title":"RStudio: Integrated Development for R","author":"RStudio Team","year":"2020"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnci\/djz079"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10549-019-05152-9"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2017170458"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1097\/RLI.0b013e3181f3fcdf"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2017161103"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2021.104364"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2016.32"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1200\/jco.2009.27.0835"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/s1470-2045(19)30275-x"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2021203633"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1525-1497.1998.cr188.x"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1552-6909.1990.tb01629.x"}],"container-title":["Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.34326","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cncr.34326","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.34326","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,12]],"date-time":"2025-10-12T18:36:26Z","timestamp":1760294186000},"score":1,"resource":{"primary":{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/10.1002\/cncr.34326"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,6]]},"references-count":34,"journal-issue":{"issue":"15","published-print":{"date-parts":[[2022,8]]}},"alternative-id":["10.1002\/cncr.34326"],"URL":"https:\/\/doi.org\/10.1002\/cncr.34326","archive":["Portico"],"relation":{},"ISSN":["0008-543X","1097-0142"],"issn-type":[{"value":"0008-543X","type":"print"},{"value":"1097-0142","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,6]]},"assertion":[{"value":"2022-03-22","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-05-10","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-01","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}