{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,19]],"date-time":"2026-10-19T16:09:33Z","timestamp":1792426173168,"version":"4.5.0"},"reference-count":44,"publisher":"Wiley","issue":"17","license":[{"start":{"date-parts":[[2020,6,18]],"date-time":"2020-06-18T00:00:00Z","timestamp":1592438400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2020,9]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Li\u2010Fraumeni syndrome (LFS) is a high\u2010risk cancer predisposition syndrome caused by pathogenic germline variants of <jats:italic>TP53<\/jats:italic>. Cancer surveillance has noted a significant survival advantage in individuals with LFS; however, little is known about the feasibility, acceptance, and psychosocial effects of such a program.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Pathogenic <jats:italic>TP53<\/jats:italic> germline variant carriers completed a 7\u2010part questionnaire evaluating sociodemographics, cancer history, surveillance participation, reasons for nonadherence, worries, and distress adapted from the Cancer Worry Scale. Counselees' common concerns and suggestions were assessed in MAXQDA Analytics Pro 12.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Forty\u2010nine participants (46 females and 3 males), aged 40.0 \u00b1 12.6\u00a0years, formed the study population; 43 (88%) had a personal cancer history (including multiple cancers in 10 [20%]). Forty\u2010three individuals participated (88%) in surveillance during the study or formerly. Willingness to undergo surveillance was influenced by satisfaction with genetic testing and counseling (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.019 [Fisher\u2010Yates test]) but not by sociodemographics, cancer history, or distress level. Almost one\u2010third of the participants reported logistical difficulties in implementing surveillance because of the high frequency of medical visits, scheduling difficulties, and the travel distance to their surveillance providers. Self\u2010reported distress and perceived emotional burden for family members and partners were moderate (median for self\u2010reported distress, 3.3; median for perceived emotional burden, 3.0). For both, the interquartile range was moderate to very high (2.7\u20103.7 and 3.0\u20103.7, respectively).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Individuals with LFS require efficient counseling as well as an accessible, well\u2010organized, interdisciplinary, standardized surveillance program to increase adherence and psychological coping.<\/jats:p><\/jats:sec>","DOI":"10.1002\/cncr.33004","type":"journal-article","created":{"date-parts":[[2020,6,18]],"date-time":"2020-06-18T11:17:39Z","timestamp":1592479059000},"page":"4032-4041","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Cancer surveillance and distress among adult pathogenic <i>TP53<\/i> germline variant carriers in Germany: A multicenter feasibility and acceptance survey"],"prefix":"10.1002","volume":"126","author":[{"given":"Nathalie","family":"Rippinger","sequence":"first","affiliation":[{"name":"Department of Gynecology and Obstetrics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine","family":"Fischer","sequence":"additional","affiliation":[{"name":"Institute of Human Genetics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1851-3747","authenticated-orcid":false,"given":"Markus W.","family":"Haun","sequence":"additional","affiliation":[{"name":"Department of General Internal Medicine and Psychosomatics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kerstin","family":"Rhiem","sequence":"additional","affiliation":[{"name":"Center for Hereditary Breast and Ovarian Cancer Center for Integrated Oncology Medical Faculty University Hospital Cologne Cologne Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sabine","family":"Grill","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Center for Hereditary Breast and Ovarian Cancer Comprehensive Cancer Center University Hospital Rechts der Isar Technical University of Munich Munich Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4237-2657","authenticated-orcid":false,"given":"Marion","family":"Kiechle","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Center for Hereditary Breast and Ovarian Cancer Comprehensive Cancer Center University Hospital Rechts der Isar Technical University of Munich Munich Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Friedrich W.","family":"Cremer","sequence":"additional","affiliation":[{"name":"SYNLAB Center for Human Genetics Mannheim Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin","family":"Kast","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Obstetrics Medical Faculty and University Hospital Carl Gustav Carus Technical University Dresden Dresden Germany"},{"name":"National Center for Tumor Diseases, Partner Site Dresden Dresden Germany"},{"name":"German Cancer Consortium and German Cancer Research Center Dresden Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huu P.","family":"Nguyen","sequence":"additional","affiliation":[{"name":"Institute of Medical Genetics and Applied Genomics University Hospital of T\u00fcbingen T\u00fcbingen Germany"},{"name":"Department of Human Genetics University of Bochum Bochum Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nina","family":"Ditsch","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Obstetrics Ludwig Maximilian University University Hospital of Munich Munich Germany"},{"name":"Department of Gynecology and Obstetrics University Hospital Augsburg Augsburg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4120-5873","authenticated-orcid":false,"given":"Christian P.","family":"Kratz","sequence":"additional","affiliation":[{"name":"Pediatric Hematology and Oncology Hannover Medical School Hannover Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julia","family":"Vogel","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Obstetrics University Hospital Charit\u00e9 Berlin Berlin Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorothee","family":"Speiser","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Adolescent Medicine Division of Pediatric Hematology and Oncology Medical Center Faculty of Medicine University of Freiburg Freiburg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1709-4448","authenticated-orcid":false,"given":"Simone","family":"Hettmer","sequence":"additional","affiliation":[{"name":"Translational Functional Cancer Genomics National Center for Tumor Diseases and German Cancer Research Center Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hanno","family":"Glimm","sequence":"additional","affiliation":[{"name":"German Cancer Consortium and German Cancer Research Center Dresden Germany"},{"name":"Translational Functional Cancer Genomics National Center for Tumor Diseases and German Cancer Research Center Heidelberg Germany"},{"name":"Department of Translational Medical Oncology National Center for Tumor Diseases, University Hospital Carl Gustav Carus, Technical University Dresden and German Cancer Research Center Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefan","family":"Fr\u00f6hling","sequence":"additional","affiliation":[{"name":"Department of Translational Medical Oncology National Center for Tumor Diseases and German Cancer Research Center Heidelberg Germany"},{"name":"German Cancer Consortium and German Cancer Research Center Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dirk","family":"J\u00e4ger","sequence":"additional","affiliation":[{"name":"German Cancer Consortium and German Cancer Research Center Heidelberg Germany"},{"name":"Department of Medical Oncology National Center for Tumor Diseases, University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephan","family":"Seitz","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Obstetrics University Medical Center Regensburg Regensburg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea","family":"Hahne","sequence":"additional","affiliation":[{"name":"BRCA Network\u2014Support for People with Hereditary Cancers"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8030-1182","authenticated-orcid":false,"given":"Imad","family":"Maatouk","sequence":"additional","affiliation":[{"name":"Department of General Internal Medicine and Psychosomatics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian","family":"Sutter","sequence":"additional","affiliation":[{"name":"Institute of Human Genetics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rita K.","family":"Schmutzler","sequence":"additional","affiliation":[{"name":"Center for Hereditary Breast and Ovarian Cancer Center for Integrated Oncology Medical Faculty University Hospital Cologne Cologne Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicola","family":"Dikow","sequence":"additional","affiliation":[{"name":"Institute of Human Genetics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1714-1147","authenticated-orcid":false,"given":"Sarah","family":"Schott","sequence":"additional","affiliation":[{"name":"Department of Gynecology and Obstetrics University Hospital Heidelberg Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,6,18]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2014.59.5728"},{"key":"e_1_2_8_3_1","first-page":"344","article-title":"Segregation analysis of cancer in families of childhood soft\u2010tissue\u2010sarcoma patients","volume":"51","author":"Lustbader ED","year":"1992","journal-title":"Am J Hum Genet"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.30248"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(16)30249-2"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.CCR-17-0408"},{"key":"e_1_2_8_7_1","unstructured":"Deutsches Konsortium Famili\u00e4rer Brust\u2010 und Eierstockkrebs.Konsensusempfehlung zum Umgang mit Ergebnissen der Multigenanalyse. Accessed June 1 2020.https:\/\/www.konsortium\u2010familiaerer\u2010brustkrebs.de\/konsensusempfehlung\/"},{"key":"e_1_2_8_8_1","volume-title":"GeneReviews","author":"Schneider K","year":"2020"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1055\/a-0574-4879"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-017-0034-6"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2017.1968"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2017.1355"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2017.1350"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2015.30"},{"key":"e_1_2_8_15_1","unstructured":"Deutsches Konsortium Famili\u00e4rer Brust\u2010 und Eierstockkrebs.Erkennung von Genver\u00e4nderungen durch einen Gentest. Accessed June 1 2020.https:\/\/www.konsortium\u2010familiaerer\u2010brustkrebs.de\/informationen\/gentest\u2010einschlusskriterien\/"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/cam4.1376"},{"key":"e_1_2_8_17_1","unstructured":"Deutsches Konsortium Famili\u00e4rer Brust\u2010 und Eierstockkrebs.Die TruRisk Gen\u2010Analyse bei famili\u00e4rem Brust\u2010 und Eierstockkrebs. Accessed June 1 2020.https:\/\/www.konsortium\u2010familiaerer\u2010brustkrebs.de\/molekulare\u2010diagnostik\/trurisk\u2010gen\u2010analyse\/"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10549-016-3748-5"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2008.01089.x"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdh318"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00404-016-4133-7"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00404-017-4376-y"},{"key":"e_1_2_8_23_1","first-page":"171","article-title":"Younger women at increased risk for breast cancer: perceived risk, psychological well\u2010being, and surveillance behavior","volume":"16","author":"Lerman C","year":"1994","journal-title":"J Natl Cancer Inst Monogr"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.2.139"},{"issue":"4","key":"e_1_2_8_25_1","first-page":"345","article-title":"Attitudes toward colon cancer gene testing: factors predicting test uptake","volume":"8","author":"Codori AM","year":"1999","journal-title":"Cancer Epidemiol Biomarkers Prev"},{"issue":"4","key":"e_1_2_8_26_1","first-page":"353","article-title":"Intention to learn results of genetic testing for hereditary colon cancer","volume":"8","author":"Vernon SW","year":"1999","journal-title":"Cancer Epidemiol Biomarkers Prev"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1191\/1478088706qp063oa"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.4135\/9781446288719"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-010-9368-z"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2019-106407"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-016-9964-7"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.critrevonc.2012.01.004"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1111\/famp.12497"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-018-0103-5"},{"key":"e_1_2_8_35_1","doi-asserted-by":"publisher","DOI":"10.1054\/bjoc.2000.1167"},{"key":"e_1_2_8_36_1","doi-asserted-by":"publisher","DOI":"10.1086\/374567"},{"key":"e_1_2_8_37_1","doi-asserted-by":"publisher","DOI":"10.1158\/0008-5472.CAN-05-4247"},{"key":"e_1_2_8_38_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2009.27.2112"},{"key":"e_1_2_8_39_1","doi-asserted-by":"publisher","DOI":"10.1002\/pon.1352"},{"key":"e_1_2_8_40_1","doi-asserted-by":"publisher","DOI":"10.1023\/A:1009495815436"},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdl069"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0033-3182(92)71993-8"},{"key":"e_1_2_8_43_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-4757-5567-1_36"},{"key":"e_1_2_8_44_1","doi-asserted-by":"publisher","DOI":"10.1002\/pon.4049"},{"key":"e_1_2_8_45_1","doi-asserted-by":"publisher","DOI":"10.1080\/07347332.2018.1543376"}],"container-title":["Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fcncr.33004","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.33004","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cncr.33004","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.33004","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,12]],"date-time":"2025-10-12T13:54:59Z","timestamp":1760277299000},"score":1,"resource":{"primary":{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/10.1002\/cncr.33004"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,6,18]]},"references-count":44,"journal-issue":{"issue":"17","published-print":{"date-parts":[[2020,9]]}},"alternative-id":["10.1002\/cncr.33004"],"URL":"https:\/\/doi.org\/10.1002\/cncr.33004","archive":["Portico"],"relation":{},"ISSN":["0008-543X","1097-0142"],"issn-type":[{"value":"0008-543X","type":"print"},{"value":"1097-0142","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,6,18]]},"assertion":[{"value":"2020-02-05","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-05-04","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-06-18","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}