{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T08:38:11Z","timestamp":1787647091841,"version":"build-2736575974"},"reference-count":56,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2022,7,14]],"date-time":"2022-07-14T00:00:00Z","timestamp":1657756800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Histopathology"],"published-print":{"date-parts":[[2022,9]]},"abstract":"<jats:sec><jats:title>Aims<\/jats:title><jats:p>SMARCB1 (INI\u20101)\u2010deficient vulvar neoplasms comprise a group of rare tumours that include epithelioid sarcoma (ES), myoepithelial carcinoma (MEC), the recently described myoepithelioma\u2010like tumour of the vulvar region (MELTVR), and sarcomas that are difficult to classify. It has been suggested that so\u2010called vulvar yolk sac tumours (YST) may represent morphologic variants of SMARCB1\u2010deficient tumours; thus, we investigated the immunoreactivity of germ cell markers in SMARCB1\u2010deficient vulvar neoplasms.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and results<\/jats:title><jats:p>Ten SMARCB1\u2010deficient vulvar neoplasms were stained with germ cell tumour markers (SALL4, glypican\u20103, OCT3\/4, and AFP) and re\u2010reviewed for morphologic features. The tumours occurred in adult females (median age 41\u2009years) and included ES (<jats:italic>n<\/jats:italic>\u2009=\u20097), MELTVR (<jats:italic>n<\/jats:italic>\u2009=\u20092), and MEC (<jats:italic>n<\/jats:italic>\u2009=\u20091). All cases showed loss of SMARCB1 expression. Four cases (40%) were focally positive for SALL4 in areas with morphology of typical\u2010appearing ES. One of these cases also showed focal staining for OCT3\/4. One ES showed a transition from typical\u2010appearing ES to YST\u2010like morphology, with diffuse expression of SALL4 and glypican\u20103, and focal expression of AFP, in these latter areas. All other tested cases were negative for AFP.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Our study reveals that SALL4, glypican\u20103, and OCT3\/4 are positive in a subset of SMARCB1\u2010deficient vulvar neoplasms, which may pose a diagnostic challenge and result in consideration of a germ cell tumour. We also highlight a case with transition from ES to YST\u2010like morphology, lending further support that YSTs of the vulva are somatically derived SMARCB1\u2010deficient neoplasms and do not represent true germ\u2010cell neoplasia.<\/jats:p><\/jats:sec>","DOI":"10.1111\/his.14709","type":"journal-article","created":{"date-parts":[[2022,6,27]],"date-time":"2022-06-27T10:18:50Z","timestamp":1656325130000},"page":"342-351","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["A Subset of <scp>SMARCB1<\/scp> (<scp>INI<\/scp>\u20101)\u2010deficient vulvar neoplasms express germ cell markers"],"prefix":"10.1111","volume":"81","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4206-2002","authenticated-orcid":false,"given":"Phoebe M","family":"Hammer","sequence":"first","affiliation":[{"name":"Department of Pathology Stanford University School of Medicine  Stanford CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David L","family":"Kolin","sequence":"additional","affiliation":[{"name":"Division of Women's and Perinatal Pathology Brigham and Women's Hospital, Harvard Medical School  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2774-2704","authenticated-orcid":false,"given":"Gregory W","family":"Charville","sequence":"additional","affiliation":[{"name":"Department of Pathology Stanford University School of Medicine  Stanford CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9178-4370","authenticated-orcid":false,"given":"W Glenn","family":"McCluggage","sequence":"additional","affiliation":[{"name":"Department of Pathology Belfast Health and Social Care Trust  Belfast UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0309-6680","authenticated-orcid":false,"given":"Brooke E","family":"Howitt","sequence":"additional","affiliation":[{"name":"Department of Pathology Stanford University School of Medicine  Stanford CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,7,14]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-pathmechdis-012418-012917"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.4161\/cbt.8.5.8019"},{"key":"e_1_2_10_4_1","first-page":"323","article-title":"The role of INI1 and the SWI\/SNF complex in the development of rhabdoid tumors: Meeting summary from the workshop on childhood atypical teratoid\/rhabdoid tumors","volume":"62","author":"Biegel JA","year":"2002","journal-title":"Cancer Res."},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2628"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2018.09.032"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000397"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/gcc.22922"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000466"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.14393"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/1746-1596-8-122"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-199702000-00002"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000001777"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.12373"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/cam4.2195"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2605.2011.01245.x"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19880801)62:3<620::AID-CNCR2820620330>3.0.CO;2-P"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gore.2015.03.006"},{"key":"e_1_2_10_19_1","first-page":"76S","article-title":"Endodermal sinus tumor of the vulva in an infant","volume":"61","author":"Dudley AG","year":"1983","journal-title":"Obstet. Gynecol."},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/0090-8258(80)90049-9"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1006\/gyno.1997.4759"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00383-012-3153-z"},{"key":"e_1_2_10_23_1","first-page":"395","article-title":"Primary yolk sac tumor (endodermal sinus tumor) of the vulva: Case report and review of the literature","volume":"27","author":"Basgul A","year":"2006","journal-title":"Eur. J. Gynaecol. Oncol."},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ygyno.2004.12.054"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/0090-8258(81)90052-4"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1048-891X.2004.14540.x"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(197804)41:4<1627::AID-CNCR2820410454>3.0.CO;2-K"},{"key":"e_1_2_10_28_1","first-page":"203","article-title":"Endodermal sinus tumour of the vulva: An interesting clinico\u2010pathological problem","volume":"19","author":"Craighead PS","year":"1993","journal-title":"Eur. J. Surg. Oncol."},{"key":"e_1_2_10_29_1","first-page":"506","article-title":"Primary endodermal sinus tumor of the vulva in a 52\u2010year\u2010old woman with long\u2010term survival: A case report","volume":"28","author":"Niwa K","year":"2007","journal-title":"Eur. J. Gynaecol. Oncol."},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.13021"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.3800310"},{"key":"e_1_2_10_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.3800934"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000001000"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41379-019-0273-1"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12105-019-01065-7"},{"key":"e_1_2_10_36_1","doi-asserted-by":"publisher","DOI":"10.1177\/1066896917741549"},{"key":"e_1_2_10_37_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0046-8177(99)90247-2"},{"key":"e_1_2_10_38_1","doi-asserted-by":"publisher","DOI":"10.3346\/jkms.1997.12.1.78"},{"key":"e_1_2_10_39_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAI.0b013e31829852e7"},{"key":"e_1_2_10_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2014.10.010"},{"key":"e_1_2_10_41_1","doi-asserted-by":"publisher","DOI":"10.1097\/IGC.0000000000001020"},{"key":"e_1_2_10_42_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41379-020-0548-6"},{"key":"e_1_2_10_43_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.14188"},{"key":"e_1_2_10_44_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2012.06.014"},{"key":"e_1_2_10_45_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000116"},{"key":"e_1_2_10_46_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41379-018-0049-z"},{"key":"e_1_2_10_47_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.2017.11"},{"key":"e_1_2_10_48_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.12460"},{"key":"e_1_2_10_49_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.13177"},{"key":"e_1_2_10_50_1","doi-asserted-by":"publisher","DOI":"10.4103\/0973-1482.63557"},{"key":"e_1_2_10_51_1","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2021-242271"},{"key":"e_1_2_10_52_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tjog.2014.08.013"},{"key":"e_1_2_10_53_1","doi-asserted-by":"publisher","DOI":"10.1002\/path.4636"},{"key":"e_1_2_10_54_1","doi-asserted-by":"publisher","DOI":"10.1002\/path.5889"},{"key":"e_1_2_10_55_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.27784"},{"key":"e_1_2_10_56_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ygyno.2007.05.045"},{"key":"e_1_2_10_57_1","doi-asserted-by":"publisher","DOI":"10.1002\/path.4362"}],"container-title":["Histopathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/his.14709","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/his.14709","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/his.14709","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,22]],"date-time":"2023-08-22T17:55:36Z","timestamp":1692726936000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/his.14709"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,7,14]]},"references-count":56,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2022,9]]}},"alternative-id":["10.1111\/his.14709"],"URL":"https:\/\/doi.org\/10.1111\/his.14709","archive":["Portico"],"relation":{},"ISSN":["0309-0167","1365-2559"],"issn-type":[{"value":"0309-0167","type":"print"},{"value":"1365-2559","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,7,14]]},"assertion":[{"value":"2022-04-07","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-05-25","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-07-14","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}