{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T17:46:22Z","timestamp":1782841582438,"version":"3.54.5"},"reference-count":18,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2016,1,14]],"date-time":"2016-01-14T00:00:00Z","timestamp":1452729600000},"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":["APMIS"],"published-print":{"date-parts":[[2016,4]]},"abstract":"<jats:p>Primary intraosseous myoepithelial tumours, including carcinomas are rare tumours. The concept of histopathological spectrum of these tumours is evolving. We describe clinicopathological and immunohistochemical features of five myoepithelial carcinomas, including molecular cytogenetic results in one case. There were five male patients within age\u2010range of 8\u201340 years (median = 26). Four tumours occurred in the long bones, including two tumours, each, in the femur and fibula, respectively, while a single tumour occurred in the proximal phalanges. Tumour size (n = 3 cases) varied from 5.6 to 8.6 cm. On radiological imaging, most tumours appeared as expansile, lytic and destructive lesions. Two tumours appeared as sclerotic lesions. Two cases were referred with diagnoses of chondrosarcomas and a single case was referred with two different diagnoses, including an adamantinoma and an osteosarcoma. Histopathological examination in all these cases showed multinodular tumours comprising mostly polygonal cells, exhibiting moderate nuclear atypia and interspersed mitotic figures within a stroma containing variable amount of myxoid, chondroid, hyalinised and osteoid\u2010like material. Three tumours revealed prominent squamous differentiation. By immunohistochemistry, tumour cells were positive for <jats:styled-content style=\"fixed-case\">EMA<\/jats:styled-content> (5\/5), pan <jats:styled-content style=\"fixed-case\">CK<\/jats:styled-content> (<jats:styled-content style=\"fixed-case\">AE<\/jats:styled-content>1\/<jats:styled-content style=\"fixed-case\">AE<\/jats:styled-content>3) (3\/3), <jats:styled-content style=\"fixed-case\">CK<\/jats:styled-content>5\/6 (4\/4), <jats:styled-content style=\"fixed-case\">CK MNF<\/jats:styled-content>116 (1\/1), S100 protein (5\/5) and <jats:styled-content style=\"fixed-case\">GFAP<\/jats:styled-content> (3\/5). The first tumour revealed <jats:italic><jats:styled-content style=\"fixed-case\">EWSR<\/jats:styled-content>1<\/jats:italic> rearrangement. The first patient, 10 months after tumour resection and a simultaneous lung metastatectomy, is free\u2010of\u2010disease (<jats:styled-content style=\"fixed-case\">FOD<\/jats:styled-content>). The second patient, 11 months after tumour resection is <jats:styled-content style=\"fixed-case\">FOD<\/jats:styled-content>. The third and fourth patients underwent wide resections and are on follow\u2010up. The fifth patient underwent resections, including a lung metastatectomy. Primary intraosseous myoepithelial carcinomas are rare and mimic conventional primary bone tumours. Some primary intraosseous myoepithelial carcinomas display <jats:italic><jats:styled-content style=\"fixed-case\">EWSR<\/jats:styled-content>1<\/jats:italic> rearrangement. Squamous differentiation may be considered as an addition to their evolving histopathological spectrum. Immunohistochemical stains constitute as a necessary tool for arriving at the correct diagnosis in such cases, which has treatment implications. Surgical resection remains the treatment mainstay.<\/jats:p>","DOI":"10.1111\/apm.12506","type":"journal-article","created":{"date-parts":[[2016,1,15]],"date-time":"2016-01-15T12:59:32Z","timestamp":1452862772000},"page":"278-290","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":26,"title":["Clinicopathological features of five unusual cases of intraosseous myoepithelial carcinomas, mimicking conventional primary bone tumours, including <i>EWSR1<\/i> rearrangement in one case"],"prefix":"10.1111","volume":"124","author":[{"given":"Bharat","family":"Rekhi","sequence":"first","affiliation":[{"name":"Department of Surgical Pathology Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sujit","family":"Joshi","sequence":"additional","affiliation":[{"name":"Department of Histopathology Deenanath Mangeshkar Hospital Pune India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yogesh","family":"Panchwagh","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Oncology Deenanath Mangeshkar Hospital Pune India"},{"name":"Sancheti Institute for Orthopaedics and Rehabilitation Pune India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashish","family":"Gulia","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology (Bone and Soft Tissues) Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita","family":"Borges","sequence":"additional","affiliation":[{"name":"Asian Institute of Oncology Mumbai India"},{"name":"SRL Diagnostics Centre of Excellence: Histopathology Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jyoti","family":"Bajpai","sequence":"additional","affiliation":[{"name":"Department of Medical Oncology Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nirmala A.","family":"Jambehekar","sequence":"additional","affiliation":[{"name":"Department of Surgical Pathology Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vinita","family":"Pant","sequence":"additional","affiliation":[{"name":"Asian Institute of Oncology Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahesh","family":"Mandholkar","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Oncology Deenanath Mangeshkar Hospital Pune India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Suman","family":"Byregowda","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology (Bone and Soft Tissues) Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ajay","family":"Puri","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology (Bone and Soft Tissues) Tata Memorial Centre Mumbai India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,1,14]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.2106\/00004623-200109000-00017"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2559.2003.01694.x"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/gcc.20819"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.prp.2011.09.004"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e3182858a0e"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.4103\/0377-4929.134706"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-200309000-00001"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0463.2007.apm_569.x"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1259\/bjr\/64670838"},{"key":"e_1_2_6_11_1","first-page":"140","article-title":"Case report 720. Multicentric mixed tumour of bone with pulmonary involvement","volume":"21","author":"Rose AG","year":"1992","journal-title":"Skeletal Radiol"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2012.06.020"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.otsr.2011.05.005"},{"key":"e_1_2_6_14_1","doi-asserted-by":"crossref","first-page":"55","DOI":"10.4137\/CPath.S582","article-title":"Squamous differentiation and cytokeratin expression in an osteosarcoma: a case report and review of the literature","volume":"1","author":"Layfield LJ","year":"2008","journal-title":"Clin Med Pathol"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000460"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1097\/BRS.0000000000000564"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e3182772a15"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/gcc.22240"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00428-012-1335-7"}],"container-title":["APMIS"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fapm.12506","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/apm.12506","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,5]],"date-time":"2023-10-05T09:07:45Z","timestamp":1696496865000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/apm.12506"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,1,14]]},"references-count":18,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2016,4]]}},"alternative-id":["10.1111\/apm.12506"],"URL":"https:\/\/doi.org\/10.1111\/apm.12506","archive":["Portico"],"relation":{},"ISSN":["0903-4641","1600-0463"],"issn-type":[{"value":"0903-4641","type":"print"},{"value":"1600-0463","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,1,14]]},"assertion":[{"value":"2015-11-12","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-12-06","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2016-01-14","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}