{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T04:34:27Z","timestamp":1791434067896,"version":"4.3.3"},"reference-count":29,"publisher":"SAGE Publications","issue":"3","license":[{"start":{"date-parts":[[2022,7,11]],"date-time":"2022-07-11T00:00:00Z","timestamp":1657497600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Int J Surg Pathol"],"published-print":{"date-parts":[[2023,5]]},"abstract":"<jats:p>\n                    Lymphangiomyomatosis is a member of the PEComa family, and usually involves the pulmonary parenchyma of middle-aged females. Infrequently, it may involve abdominal and retroperitoneal lymph nodes, and rarely it has been described to be associated with fallopian tube-type ciliated epithelium co-existing in one neoplasm. To increase our understanding of this unusual tumor, we describe the morphology and genetics of one case and review the literature. We present the case of a 50-year-old female found to have 12.5 and 7.7\u2005cm cystic retroperitoneal masses, describe its unique pathological features and review the literature on the previously reported cases. Based on its unique morphological, immunohistochemical, and molecular features we propose the term adenoPEComa to represent this entity. This case represents a rare example of adenoPEComa with lymphangiomyomatosis of the lymph nodes. It is the first example that has undergone next-generation sequencing revealing a mutation in\n                    <jats:italic toggle=\"yes\">TSC2<\/jats:italic>\n                    making it a confirmed member of the PEComa family of tumors.\n                  <\/jats:p>","DOI":"10.1177\/10668969221105616","type":"journal-article","created":{"date-parts":[[2022,7,11]],"date-time":"2022-07-11T07:44:06Z","timestamp":1657525446000},"page":"332-337","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":1,"title":["Retroperitoneal Cystic Adenopecoma with Genetic Analysis: A Rare Neoplasm"],"prefix":"10.1177","volume":"31","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8827-6794","authenticated-orcid":false,"given":"Domenika","family":"Ortiz Requena","sequence":"first","affiliation":[{"name":"Jackson Memorial Hospital\/University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Belen","family":"Goiburu","sequence":"additional","affiliation":[{"name":"Jackson Memorial Hospital\/University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaylou","family":"Velez-Torres","sequence":"additional","affiliation":[{"name":"University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nemencio","family":"Ronquillo","sequence":"additional","affiliation":[{"name":"University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alan S.","family":"Livingstone","sequence":"additional","affiliation":[{"name":"University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew E.","family":"Rosenberg","sequence":"additional","affiliation":[{"name":"University of Miami Miller School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2022,7,11]]},"reference":[{"issue":"5","key":"e_1_3_3_2_2","doi-asserted-by":"crossref","first-page":"e0232487","DOI":"10.1371\/journal.pone.0232487","article-title":"Prevalence of endosalpingiosis and other benign gynecologic lesions","volume":"15","author":"Sunde J","unstructured":"Sunde J, Wasickanin M, Katz TA, et al. Prevalence of endosalpingiosis and other benign gynecologic lesions. PLoS One. 2020 May 13;15(5):e0232487. Erratum in: PLoS One. 2021 Mar 4;16(3):e0248443. PMID: 32401810; PMCID: PMC7219775.","journal-title":"PLoS One"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1097\/MD.0000000000008336"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.anndiagpath.2015.06.003"},{"issue":"1","key":"e_1_3_3_5_2","first-page":"33","article-title":"Diagnostic management of occult nodal lymphangioleiomyomatosis detected during pelvic cancer staging. Localized finding or systemic disease?","volume":"36","author":"Remo A","year":"2019","unstructured":"Remo A, Zanella C, Parcesepe P, et al. Diagnostic management of occult nodal lymphangioleiomyomatosis detected during pelvic cancer staging. Localized finding or systemic disease?. Sarcoidosis Vasc Diffuse Lung Dis. 2019;36(1):33\u201038.","journal-title":"Sarcoidosis Vasc Diffuse Lung Dis"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.273.48.32347"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e31824a8a37"},{"key":"e_1_3_3_8_2","doi-asserted-by":"publisher","DOI":"10.1055\/s-0032-1325159"},{"key":"e_1_3_3_9_2","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.164.4.2101154"},{"issue":"6","key":"e_1_3_3_10_2","doi-asserted-by":"crossref","first-page":"2138","DOI":"10.1164\/ajrccm.152.6.8520787","article-title":"How common are renal angiomyolipomas in patients with pulmonary lymphangiomyomatosis?","volume":"152","author":"Bernstein SM","unstructured":"Bernstein SM, Newell JDJr, Adamczyk D, Mortenson RL, King TEJr, Lynch DA. How common are renal angiomyolipomas in patients with pulmonary lymphangiomyomatosis? Am J Respir Crit Care Med. 1995 Dec;152(6 Pt 1):2138\u20102143. PMID: 8520787.","journal-title":"Am J Respir Crit Care Med"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1378\/chest.115.4.1041"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2559.2005.02316.x"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.55418\/1881041794"},{"issue":"2","key":"e_1_3_3_14_2","first-page":"348","article-title":"Pulmonary lymphangiomyomatosis","volume":"79","author":"Corrin B","year":"1975","unstructured":"Corrin B, Liebow AA, Friedman PJ. Pulmonary lymphangiomyomatosis. A Review Am J Pathol. 1975;79(2):348\u2010382.","journal-title":"A Review Am J Pathol"},{"key":"e_1_3_3_15_2","doi-asserted-by":"publisher","DOI":"10.1053\/hupa.2000.18500"},{"key":"e_1_3_3_16_2","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000416"},{"key":"e_1_3_3_17_2","doi-asserted-by":"publisher","DOI":"10.1177\/106689690100900212"},{"key":"e_1_3_3_18_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.277.5327.805"},{"key":"e_1_3_3_19_2","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000349"},{"key":"e_1_3_3_20_2","doi-asserted-by":"publisher","DOI":"10.5858\/2008-0542-RAR1.1"},{"key":"e_1_3_3_21_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI58709"},{"key":"e_1_3_3_22_2","doi-asserted-by":"publisher","DOI":"10.1183\/20734735.0007-2020"},{"key":"e_1_3_3_23_2","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M202678200"},{"key":"e_1_3_3_24_2","doi-asserted-by":"publisher","DOI":"10.1007\/s12010-017-2657-3"},{"key":"e_1_3_3_25_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0002-9378(16)42561-5"},{"key":"e_1_3_3_26_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.tjog.2019.03.006"},{"key":"e_1_3_3_27_2","doi-asserted-by":"publisher","DOI":"10.4076\/1757-1626-2-6331"},{"key":"e_1_3_3_28_2","doi-asserted-by":"publisher","DOI":"10.1080\/00313020903041069"},{"key":"e_1_3_3_29_2","doi-asserted-by":"publisher","DOI":"10.2214\/AJR.08.1174"},{"key":"e_1_3_3_30_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0463.2007.00668.x"}],"container-title":["International Journal of Surgical Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10668969221105616","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/10668969221105616","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10668969221105616","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T15:54:58Z","timestamp":1787759698000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/10668969221105616"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,7,11]]},"references-count":29,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,5]]}},"alternative-id":["10.1177\/10668969221105616"],"URL":"https:\/\/doi.org\/10.1177\/10668969221105616","relation":{},"ISSN":["1066-8969","1940-2465"],"issn-type":[{"value":"1066-8969","type":"print"},{"value":"1940-2465","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,7,11]]}}}