{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,14]],"date-time":"2025-10-14T00:05:15Z","timestamp":1760400315451,"version":"build-2065373602"},"reference-count":32,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2025,8,29]],"date-time":"2025-08-29T00:00:00Z","timestamp":1756425600000},"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":["J Cutan Pathol"],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:p>Hidradenoma can exhibit several cell types, including clear cells, polyhedral eosinophilic cells, squamoid cells, mucinous cells, oxyphilic (oncocytic) cells, and transitional (intermediate) cells. However, sebocytes have not yet been described in hidradenoma. Here, we present a case of <jats:italic>CRTC1::MAML2<\/jats:italic>\u2010positive hidradenoma with sebaceous differentiation. In addition, this tumor had two cell\u2010type layers that were morphologically similar to apocrine glandular cells and myoepithelial cells throughout the lesion. A 66\u2010year\u2010old male presented with a 20\u2009mm nodule on his upper lip with a 3\u2010year history. Histopathological examination revealed a multinodular tumor adherent to the overlying epidermis. No surrounding salivary glands were seen. The tumor consisted of eosinophilic ductoglandular cells surrounded by basaloid cells at the outer periphery. Numerous sebocytes were scattered throughout the tumor. Severe nuclear atypia or mitotic figures were not observed. Immunohistochemical examination showed no expression of \u03b1\u2010smooth muscle actin, calponin, and S100 protein in the tumor cells. Adipophilin highlighted scattered sebocytic tumor cells. Immunoexpression of MLH1, PMS2, MSH2, and MSH6 was preserved. <jats:italic>MAML2<\/jats:italic> break\u2010apart fluorescence in\u00a0situ hybridization revealed frequent split signals. Sanger sequencing revealed a <jats:italic>CRTC1<\/jats:italic>(e1)<jats:italic>::MAML2<\/jats:italic>(e2) fusion. Based on this report, hidradenoma should be included in the differential diagnosis of cutaneous adnexal tumors with sebaceous differentiation other than sebaceous adnexal tumors. In addition, sebocytes should be added to the list of tumor cell types in hidradenoma.<\/jats:p>","DOI":"10.1111\/cup.14862","type":"journal-article","created":{"date-parts":[[2025,8,30]],"date-time":"2025-08-30T06:11:18Z","timestamp":1756534278000},"page":"690-695","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["<scp><i>CRTC1<\/i><\/scp>::<scp><i>MAML2<\/i><\/scp>\u2010Positive Hidradenoma With Sebaceous Differentiation"],"prefix":"10.1111","volume":"52","author":[{"given":"Megumi","family":"Aoki","sequence":"first","affiliation":[{"name":"Department of Dermato\u2010Oncology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4165-1809","authenticated-orcid":false,"given":"Keisuke","family":"Goto","sequence":"additional","affiliation":[{"name":"Department of Dermato\u2010Oncology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"},{"name":"Department of Anatomic Pathology Tokyo Medical University  Tokyo Japan"},{"name":"Department of Pathology Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital  Tokyo Japan"},{"name":"Department of Diagnostic Pathology Shizuoka Cancer Center Hospital  Sunto Japan"},{"name":"Department of Diagnostic Pathology Chutoen General Medical Center  Kakegawa Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6482-8953","authenticated-orcid":false,"given":"Kazuyasu","family":"Fujii","sequence":"additional","affiliation":[{"name":"Department of Dermato\u2010Oncology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"},{"name":"Department of Dermatology Kindai University Hospital  Osaka Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mitsuharu","family":"Nomoto","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Pathology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0943-4115","authenticated-orcid":false,"given":"Masamichi","family":"Goto","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Pathology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Manami","family":"Kajiwara","sequence":"additional","affiliation":[{"name":"Department of Anatomic Pathology Tokyo Medical University  Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2075-9738","authenticated-orcid":false,"given":"Toshitaka","family":"Nagao","sequence":"additional","affiliation":[{"name":"Department of Anatomic Pathology Tokyo Medical University  Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2001-5341","authenticated-orcid":false,"given":"Shigeto","family":"Matsushita","sequence":"additional","affiliation":[{"name":"Department of Dermato\u2010Oncology National Hospital Organization Kagoshima Medical Center  Kagoshima Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,8,29]]},"reference":[{"key":"e_1_2_7_2_1","first-page":"449","article-title":"Hidradenoma und adenoma hidradenoides","volume":"6","author":"Pick L.","year":"1902","journal-title":"Virchows Archiv"},{"key":"e_1_2_7_3_1","first-page":"548","article-title":"Zur histologie der hidroadenome","volume":"55","author":"Mayer I.","year":"1941","journal-title":"Frankfurter Zeitschrift f\u00fcr Pathologie"},{"key":"e_1_2_7_4_1","first-page":"9","volume-title":"Cutaneous Adnexal Tumors","author":"Kazakov D. 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