{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,29]],"date-time":"2025-08-29T10:15:55Z","timestamp":1756462555605,"version":"3.44.0"},"reference-count":11,"publisher":"BMJ","issue":"11","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2019,9,26]]},"published-print":{"date-parts":[[2019,11]]},"abstract":"<jats:p>We report a unique uterine neoplasm, favoured to represent an isolated extrapulmonary lymphangioleiomyoma with unusual pathological features, in a postmenopausal woman without tuberous sclerosis complex. The large neoplasm consisted of smooth muscle fascicles and cystic spaces lined by lymphatic cells, which were negative for the melanocytic staining that is characteristically positive in lymphangioleiomyomatosis (LAM). There are fewer than 30 cases of uterine LAM reported, none of which have demonstrated this morphology or these immunohistochemical findings. The origin of LAM cells in the more typical pulmonary LAM remains unclear; the unusual features in this case may represent a distinct pathological entity or a rare variant of typical extrapulmonary LAM, and may contribute to determining the cellular origin of these rare tumours. Conversely, this may represent a case of \u2018prepulmonary\u2019 LAM, providing supporting evidence for a possible gynaecological origin of these tumours in the broader affected (almost exclusively female) population.<\/jats:p>","DOI":"10.1136\/bcr-2019-231683","type":"journal-article","created":{"date-parts":[[2019,11,8]],"date-time":"2019-11-08T09:39:51Z","timestamp":1573205991000},"page":"e231683","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Isolated sporadic uterine lymphangioleiomyoma with unusual clinical and pathological features"],"prefix":"10.1136","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2731-1496","authenticated-orcid":false,"given":"Sebastian","family":"Leathersich","sequence":"first","affiliation":[{"name":"Department of Obstetrics and Gynaecology, King Edward Memorial Hospital for Women, Subiaco, Western Australia, Australia"},{"name":"Department of Obstetrics and Gynaecology, Armadale Health Service, Armadale, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mei Hui Eleanor","family":"Koay","sequence":"additional","affiliation":[{"name":"Department of Anatomical Pathology, PathWest Laboratory Medicine Western Australia, Subiaco, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alireza","family":"Khani","sequence":"additional","affiliation":[{"name":"Department of Anatomical Pathology, PathWest Laboratory Medicine Western Australia, Subiaco, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sangeeta","family":"Malla Bhat","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynaecology, Armadale Health Service, Armadale, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2019,11,7]]},"reference":[{"key":"2025082711065954000_12.11.e231683.1","doi-asserted-by":"crossref","first-page":"085","DOI":"10.1055\/s-2002-25298","article-title":"Lymphangioleiomyomatosis","volume":"23","author":"Johnson","year":"2002","journal-title":"Semin Respir Crit Care Med"},{"key":"2025082711065954000_12.11.e231683.2","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.164.4.2101154"},{"key":"2025082711065954000_12.11.e231683.3","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2009.05.011"},{"key":"2025082711065954000_12.11.e231683.4","doi-asserted-by":"publisher","DOI":"10.1136\/jclinpath-2015-202945"},{"key":"2025082711065954000_12.11.e231683.5","doi-asserted-by":"crossref","first-page":"1823","DOI":"10.5858\/2009-0576-RS.1","article-title":"Pulmonary lymphangioleiomyomatosis","volume":"134","author":"Zhang","year":"2010","journal-title":"Arch Pathol Lab Med"},{"key":"2025082711065954000_12.11.e231683.6","first-page":"3353","article-title":"Immunohistochemical evaluation of pulmonary lymphangioleiomyomatosis","volume":"35","author":"Grzegorek","year":"2015","journal-title":"Anticancer Res"},{"key":"2025082711065954000_12.11.e231683.7","doi-asserted-by":"crossref","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":"2025082711065954000_12.11.e231683.8","doi-asserted-by":"publisher","DOI":"10.1210\/me.2013-1059"},{"key":"2025082711065954000_12.11.e231683.9","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e318235edbd"},{"key":"2025082711065954000_12.11.e231683.10","doi-asserted-by":"crossref","first-page":"1015","DOI":"10.1097\/PAS.0000000000000416","article-title":"Incidental pelvic and para-aortic lymph node lymphangioleiomyomatosis detected during surgical staging of pelvic cancer in women without symptomatic pulmonary lymphangioleiomyomatosis or tuberous sclerosis complex","volume":"39","author":"Rabban","year":"2015","journal-title":"Am J Surg Pathol"},{"key":"2025082711065954000_12.11.e231683.11","doi-asserted-by":"crossref","first-page":"879","DOI":"10.5858\/2003-127-879-LRLMM","article-title":"Localized retroperitoneal lymphangioleiomyomatosis mimicking malignancy. A case report and review of the literature","volume":"127","author":"Jaiswal","year":"2003","journal-title":"Arch Pathol Lab Med"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2019-231683","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T18:07:10Z","timestamp":1756318030000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2019-231683"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,11]]},"references-count":11,"journal-issue":{"issue":"11","published-online":{"date-parts":[[2019,11,7]]},"published-print":{"date-parts":[[2019,11]]}},"alternative-id":["10.1136\/bcr-2019-231683"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2019-231683","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2019,11]]}}}