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Extramammary Paget disease (EMPD) of the oral cavity is exceedingly rare, with only eight reported cases, four of which were associated with an underlying internal malignancy. Here, we report a case of oral EMPD involving the buccal mucosa and gingiva of an 81\u2010year\u2010old male with no known underlying internal malignancy. The Paget cells were positive for CK7, CK20, CAM5.2, and androgen receptor, but negative for SOX10 and p63. The immunophenotype, association with internal malignancies, and treatment approaches for oral EMPD are reviewed.<\/jats:p>","DOI":"10.1111\/cup.14663","type":"journal-article","created":{"date-parts":[[2024,5,30]],"date-time":"2024-05-30T05:20:13Z","timestamp":1717046413000},"page":"666-671","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Unusual case of extramammary Paget disease of the oral mucosa: A case report and literature review"],"prefix":"10.1111","volume":"51","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9191-5932","authenticated-orcid":false,"given":"Grant","family":"Eilers","sequence":"first","affiliation":[{"name":"Department of Pathology University of California, San Francisco  San Francisco California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Richard C.","family":"Jordan","sequence":"additional","affiliation":[{"name":"Department of Pathology University of California, San Francisco  San Francisco California USA"},{"name":"UCSF Dermatopathology &amp; Oral Pathology Service University of California, San Francisco  San Francisco California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,5,29]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.3322\/canjclin.21.5.303"},{"issue":"3","key":"e_1_2_5_3_1","first-page":"291","article-title":"Mammary Paget's disease and associated carcinoma. 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