{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T01:57:49Z","timestamp":1788573469141,"version":"build-2803163510"},"reference-count":19,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2015,5,12]],"date-time":"2015-05-12T00:00:00Z","timestamp":1431388800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Cutan Pathol"],"published-print":{"date-parts":[[2015,6]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Multiple <jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content> negative melanocytic neoplasms are a hallmark of familial cancer susceptibility syndrome caused by <jats:italic><jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content><\/jats:italic> germline mutation. The syndrome is characterized by increased incidence of renal cell carcinoma, mesothelioma, cholangiocarcinoma, cutaneous and uveal melanoma and some other neoplasms.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We report histomorphologic characteristics of six cutaneous melanocytic neoplasms with loss of <jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content> expression in two members of a family with <jats:italic><jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content><\/jats:italic>\u2010associated cancer susceptibility syndrome.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The neoplasms were dermal melanocytic nevi characterized by a proliferation of large epithelioid (spitzoid) melanocytes, and adipocytic metaplasia. Nuclear pseudoinclusions and multinucleated melanocytes were present in most neoplasms. In two of the cases, a nodular melanoma was found associated with a dermal nevus. None of the melanomas recurred or metastasized after 6 and 3\u2009years of follow up.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>We report two new cases of melanoma arising in a <jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content>\u2010deficient melanocytic nevus in the setting of familial tumor predisposition syndrome. Adipocytic metaplasia and nuclear pseudoinclusions may be additional morphologic clues to a <jats:styled-content style=\"fixed-case\">BAP1<\/jats:styled-content>\u2010deficient nevus. It remains to be seen whether these features are more common in familial than sporadic lesions.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cup.12493","type":"journal-article","created":{"date-parts":[[2015,4,23]],"date-time":"2015-04-23T06:33:02Z","timestamp":1429770782000},"page":"406-412","source":"Crossref","is-referenced-by-count":30,"title":["Histomorphologic spectrum of BAP1 negative melanocytic neoplasms in a family with <i>BAP1<\/i>\u2010associated cancer susceptibility syndrome"],"prefix":"10.1111","volume":"42","author":[{"given":"Zlatko","family":"Maru\u0161i\u0107","sequence":"first","affiliation":[{"name":"Ljudevit Jurak Department of Pathology Clinical Hospital Center \u201cSestre milosrdnice\u201d  Zagreb Croatia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marija","family":"Buljan","sequence":"additional","affiliation":[{"name":"Department of Dermatology Clinical Hospital Center \u201cSestre milosrdnice\u201d  Zagreb Croatia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Klaus J.","family":"Busam","sequence":"additional","affiliation":[{"name":"Department of Pathology Memorial Sloan Kettering Cancer Center  New York NY USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,5,12]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.910"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.912"},{"key":"e_1_2_5_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.04.012"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2011-100156"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0035295"},{"key":"e_1_2_5_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/gcc.22129"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2323"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.855"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1194472"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e3182498be5"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e318263648c"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000209"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.CCR-11-2375"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2013.10.032"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.1136\/jcp.48.7.676"},{"key":"e_1_2_5_17_1","doi-asserted-by":"publisher","DOI":"10.1097\/DAD.0b013e3181e448cb"},{"key":"e_1_2_5_18_1","doi-asserted-by":"publisher","DOI":"10.1097\/00000372-198308000-00011"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1158\/0008-5472.CAN-05-1343"},{"key":"e_1_2_5_20_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2011.41.2965"}],"container-title":["Journal of Cutaneous Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fcup.12493","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cup.12493","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,3]],"date-time":"2023-10-03T04:23:10Z","timestamp":1696306990000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cup.12493"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,5,12]]},"references-count":19,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2015,6]]}},"alternative-id":["10.1111\/cup.12493"],"URL":"https:\/\/doi.org\/10.1111\/cup.12493","archive":["Portico"],"relation":{},"ISSN":["0303-6987","1600-0560"],"issn-type":[{"value":"0303-6987","type":"print"},{"value":"1600-0560","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,5,12]]}}}