{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,6]],"date-time":"2026-11-06T17:54:08Z","timestamp":1793987648540,"version":"4.8.0"},"reference-count":62,"publisher":"Oxford University Press (OUP)","issue":"7","license":[{"start":{"date-parts":[[2025,5,8]],"date-time":"2025-05-08T00:00:00Z","timestamp":1746662400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"name":"JSPS\/MEXT KAKENHI","award":["22K09421"],"award-info":[{"award-number":["22K09421"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,7,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Perivascular epithelioid cell tumors (PEComas) are a rare family of mesenchymal tumors that includes angiomyolipoma, lymphangioleiomyomatosis, pulmonary clear cell \u201csugar\u201d tumors, and PEComa-not otherwise specified. This study aimed to provide a comprehensive review of the clinical features, molecular biology, and current status of PEComa treatment. It reportedly occurs at several sites, including the uterus, kidney, liver, lung, abdominopelvic soft tissue, gastrointestinal organs, retroperitoneum, soft tissue, bone, and skin. More common in women, it occurs in young to middle-aged people. Although the disease generally follows a benign course, cases of malignant PEComa have been reported. Malignant PEComa is characterized by a large tumor size, a high mitotic rate, and the presence of necrosis and nuclear atypia. Immunohistochemically, PEComas typically express melanocytic markers such as human melanoma black 45 (HMB45) and melanoma antigen (melan-A) and muscle markers such as smooth muscle actin (\u03b1-SMA), desmin, and caldesmon. More recently, a subtype of PEComa harboring TFE3 gene rearrangement that is mutually exclusive with tuberous sclerosis complex (TSC) mutations has been identified. The identification of TFE3 gene rearrangement can help confirm the diagnosis. The distinctive features of these TFE3-rearranged PEComas include a young-age tendency, the absence of an association with tuberous sclerosis, predominant alveolar architecture and epithelioid cytology, minimal immunoreactivity for muscle markers, and strong (3+) TFE3 immunoreactivity. Surgery is the curative treatment of choice; however, there are reports of cases and randomized controlled trials showing the efficacy of mTOR inhibitors. To the best of our knowledge, there are no reports of radiation therapy\u2019s efficacy.<\/jats:p>","DOI":"10.1093\/jjco\/hyaf056","type":"journal-article","created":{"date-parts":[[2025,3,23]],"date-time":"2025-03-23T07:53:53Z","timestamp":1742716433000},"page":"691-698","source":"Crossref","is-referenced-by-count":23,"title":["PEComa\u2014its clinical features, histopathology, and current therapy"],"prefix":"10.1093","volume":"55","author":[{"given":"Yuya","family":"Izubuchi","sequence":"first","affiliation":[{"name":"Department of Orthopaedics and Rehabilitaion Medicine, Unit of Surgery, Division of Medicine, Faculty of Medical Sciences , University of Fukui 23-3, Matsuokashimoaizuki, Eiheiji-cho, Yoshida-gun, Fukui 910-1193 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takaaki","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics and Rehabilitaion Medicine, Unit of Surgery, Division of Medicine, Faculty of Medical Sciences , University of Fukui 23-3, Matsuokashimoaizuki, Eiheiji-cho, Yoshida-gun, Fukui 910-1193 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2025,5,8]]},"reference":[{"key":"2025070702413543500_ref1","doi-asserted-by":"publisher","first-page":"307","DOI":"10.1097\/00000478-199203000-00013","article-title":"PEC and sugar","volume":"16","author":"Bonetti","year":"1992","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref2","volume-title":"International academy of P","author":"Fletcher","year":"2002"},{"key":"2025070702413543500_ref3","doi-asserted-by":"publisher","first-page":"1558","DOI":"10.1097\/01.pas.0000173232.22117.37","article-title":"Perivascular epithelioid cell neoplasms of soft tissue and gynecologic origin: A clinicopathologic study of 26 cases and review of the literature","volume":"29","author":"Folpe","year":"2005","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref4","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1111\/j.1365-2559.2005.02316.x","article-title":"PEComa: What do we know so far?","volume":"48","author":"Hornick","year":"2006","journal-title":"Histopathology"},{"key":"2025070702413543500_ref5","doi-asserted-by":"publisher","first-page":"648","DOI":"10.5858\/133.4.648","article-title":"Perivascular epithelioid cell tumor","volume":"133","author":"Armah","year":"2009","journal-title":"Arch Pathol Lab Med"},{"key":"2025070702413543500_ref6","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1016\/j.humpath.2009.05.011","article-title":"Perivascular epithelioid cell neoplasms: Pathology and pathogenesis","volume":"41","author":"Folpe","year":"2010","journal-title":"Hum Pathol"},{"key":"2025070702413543500_ref7","doi-asserted-by":"publisher","first-page":"1239","DOI":"10.1097\/00000478-200009000-00007","article-title":"Clear cell myomelanocytic tumor of the falciform ligament\/ligamentum teres: A novel member of the perivascular epithelioid clear cell family of tumors with a predilection for children and young adults","volume":"24","author":"Folpe","year":"2000","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref8","doi-asserted-by":"publisher","first-page":"1769","DOI":"10.1097\/PAS.0b013e31829caab3","article-title":"PEComa of the gastrointestinal tract: Clinicopathologic study of 35 cases with evaluation of prognostic parameters","volume":"37","author":"Doyle","year":"2013","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref9","doi-asserted-by":"publisher","first-page":"493","DOI":"10.1097\/PAS.0b013e318161dc34","article-title":"Sclerosing PEComa: Clinicopathologic analysis of a distinctive variant with a predilection for the retroperitoneum","volume":"32","author":"Hornick","year":"2008","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref10","doi-asserted-by":"publisher","first-page":"1370","DOI":"10.1097\/PAS.0000000000001119","article-title":"Uterine PEComas: A morphologic, immunohistochemical, and molecular analysis of 32 Tumors","volume":"42","author":"Bennett","year":"2018","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref11","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1097\/00000478-200201000-00001","article-title":"Perivascular epithelioid cell tumor ('PEComa') of the uterus: A subset of HMB-45-positive epithelioid mesenchymal neoplasms with an uncertain relationship to pure smooth muscle tumors","volume":"26","author":"Vang","year":"2002","journal-title":"Am J Surg Pathol"},{"key":"2025070702413543500_ref12","doi-asserted-by":"publisher","first-page":"498","DOI":"10.1111\/j.1365-2559.2005.02105.x","article-title":"Cutaneous clear cell myomelanocytic tumour: A new member of the growing family of perivascular epithelioid cell tumours (PEComas). 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