{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T18:42:28Z","timestamp":1789670548031,"version":"4.0.1"},"reference-count":29,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-017"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-037"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-012"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-029"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-004"}],"funder":[{"DOI":"10.13039\/100016453","name":"Department of Pathology, Johns Hopkins University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016453","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.fr","clinicalkey.jp","clinicalkey.com.au","clinicalkey.es","clinicalkey.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Human Pathology"],"published-print":{"date-parts":[[2025,3]]},"DOI":"10.1016\/j.humpath.2025.105764","type":"journal-article","created":{"date-parts":[[2025,4,3]],"date-time":"2025-04-03T13:03:02Z","timestamp":1743685382000},"page":"105764","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":4,"special_numbering":"C","title":["Examination of the histogenesis of epithelial cysts in angiomyolipoma with epithelial cysts (AMLEC) using GPNMB and TSC2 immunostains"],"prefix":"10.1016","volume":"157","author":[{"given":"Huili","family":"Li","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oluwademilade","family":"Dairo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tamara","family":"Lotan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pedram","family":"Argani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0990-1115","authenticated-orcid":false,"given":"Andres","family":"Matoso","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.humpath.2025.105764_bib1","doi-asserted-by":"crossref","first-page":"1469","DOI":"10.1097\/PAS.0b013e318299f2d1","article-title":"The international society of urological pathology (ISUP) vancouver classification of renal neoplasia","volume":"37","author":"Srigley","year":"2013","journal-title":"Am J Surg Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib2","doi-asserted-by":"crossref","first-page":"593","DOI":"10.1097\/01.pas.0000194298.19839.b4","article-title":"Angiomyolipoma with epithelial cysts (AMLEC): a distinct cystic variant of angiomyolipoma","volume":"30","author":"Fine","year":"2006","journal-title":"Am J Surg Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib3","doi-asserted-by":"crossref","first-page":"1470","DOI":"10.1016\/j.mayocp.2020.11.004","article-title":"Renal neoplasia in tuberous sclerosis: a study of 41 patients","volume":"96","author":"Gupta","year":"2021","journal-title":"Mayo Clin Proc"},{"key":"10.1016\/j.humpath.2025.105764_bib4","doi-asserted-by":"crossref","first-page":"853","DOI":"10.1111\/j.1464-410X.2004.05046.x","article-title":"An epidemiological study of renal pathology in tuberous sclerosis complex","volume":"94","author":"O'Callaghan","year":"2004","journal-title":"BJU Int"},{"key":"10.1016\/j.humpath.2025.105764_bib5","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1006242","article-title":"Whole exome sequencing identifies TSC1\/TSC2 biallelic loss as the primary and sufficient driver event for renal angiomyolipoma development","volume":"12","author":"Giannikou","year":"2016","journal-title":"PLoS Genet"},{"key":"10.1016\/j.humpath.2025.105764_bib6","doi-asserted-by":"crossref","DOI":"10.3390\/cancers15164043","article-title":"Recent advances in renal tumors with TSC\/mTOR pathway abnormalities in patients with tuberous sclerosis complex and in the sporadic setting","volume":"15","author":"Kapur","year":"2023","journal-title":"Cancers"},{"key":"10.1016\/j.humpath.2025.105764_bib7","doi-asserted-by":"crossref","first-page":"1361","DOI":"10.1016\/j.humpath.2007.01.028","article-title":"Activation of the mTOR pathway in sporadic angiomyolipomas and other perivascular epithelioid cell neoplasms","volume":"38","author":"Kenerson","year":"2007","journal-title":"Hum Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib8","doi-asserted-by":"crossref","first-page":"100","DOI":"10.1038\/modpathol.2011.136","article-title":"Cathepsin K expression in the spectrum of perivascular epithelioid cell (PEC) lesions of the kidney","volume":"25","author":"Martignoni","year":"2012","journal-title":"Mod Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib9","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0024919","article-title":"Angiomyolipoma have common mutations in TSC2 but no other common genetic events","volume":"6","author":"Qin","year":"2011","journal-title":"PLoS One"},{"key":"10.1016\/j.humpath.2025.105764_bib10","doi-asserted-by":"crossref","DOI":"10.3389\/fonc.2024.1274953","article-title":"TSC2 nonsense mutation in angiomyolipoma with epithelial cysts: a case report and literature review","volume":"14","author":"Song","year":"2024","journal-title":"Front Oncol"},{"key":"10.1016\/j.humpath.2025.105764_bib11","article-title":"TSC1 splicing mutation in renal angiomyolipoma with epithelial cysts without fat: a very rare case report and literature review","volume":"10","author":"Xu","year":"2024","journal-title":"Heliyon"},{"key":"10.1016\/j.humpath.2025.105764_bib12","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-017-16988-w","article-title":"Mutational analysis of TSC1 and TSC2 genes in tuberous sclerosis complex patients from Greece","volume":"7","author":"Avgeris","year":"2017","journal-title":"Sci Rep"},{"key":"10.1016\/j.humpath.2025.105764_bib13","doi-asserted-by":"crossref","first-page":"8313","DOI":"10.1074\/jbc.C500451200","article-title":"TSC1 stabilizes TSC2 by inhibiting the interaction between TSC2 and the HERC1 ubiquitin ligase","volume":"281","author":"Chong-Kopera","year":"2006","journal-title":"J Biol Chem"},{"key":"10.1016\/j.humpath.2025.105764_bib14","doi-asserted-by":"crossref","first-page":"87","DOI":"10.1111\/j.1749-6632.2009.05117.x","article-title":"The tuberous sclerosis complex","volume":"1184","author":"Orlova","year":"2010","journal-title":"Ann N Y Acad Sci"},{"key":"10.1016\/j.humpath.2025.105764_bib15","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1002\/path.5875","article-title":"GPNMB expression identifies TSC1\/2\/mTOR-associated and MiT family translocation-driven renal neoplasms","volume":"257","author":"Salles","year":"2022","journal-title":"J Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib16","doi-asserted-by":"crossref","first-page":"4245","DOI":"10.1038\/s41467-021-24499-6","article-title":"TSC2 regulates lysosome biogenesis via a non-canonical RAGC and TFEB-dependent mechanism","volume":"12","author":"Alesi","year":"2021","journal-title":"Nat Commun"},{"key":"10.1016\/j.humpath.2025.105764_bib17","doi-asserted-by":"crossref","first-page":"1267","DOI":"10.1097\/PAS.0000000000002117","article-title":"Positive GPNMB immunostaining differentiates renal cell carcinoma with fibromyomatous stroma associated with TSC1\/2\/MTOR alterations from others","volume":"47","author":"Li","year":"2023","journal-title":"Am J Surg Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib18","doi-asserted-by":"crossref","first-page":"2147","DOI":"10.1158\/1078-0432.CCR-09-1611","article-title":"Glycoprotein nonmetastatic B is an independent prognostic indicator of recurrence and a novel therapeutic target in breast cancer","volume":"16","author":"Rose","year":"2010","journal-title":"Clin Cancer Res"},{"key":"10.1016\/j.humpath.2025.105764_bib19","doi-asserted-by":"crossref","first-page":"102","DOI":"10.1016\/j.steroids.2017.10.013","article-title":"Glycoprotein non-metastatic melanoma protein B (GPNMB) and cancer: a novel potential therapeutic target","volume":"133","author":"Taya","year":"2018","journal-title":"Steroids"},{"key":"10.1016\/j.humpath.2025.105764_bib20","doi-asserted-by":"crossref","first-page":"669","DOI":"10.1038\/modpathol.3800572","article-title":"Cystic angiomyolipoma of the kidney: a clinicopathologic description of 11 cases","volume":"19","author":"Davis","year":"2006","journal-title":"Mod Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib21","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1016\/j.anndiagpath.2011.03.004","article-title":"Renal epithelioid angiomyolipoma with epithelial cysts: demonstration of Melan A and HMB45 positivity in the cystic epithelial lining","volume":"16","author":"Filho Jdo","year":"2012","journal-title":"Ann Diagn Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib22","article-title":"Immunophenotypic analysis of angiomyolipoma with epithelial cysts, comparison to mixed epithelial and stromal tumors and epithelial and stromal elements of normal kidney and ovaries","author":"Kilic","year":"2024","journal-title":"Virchows Arch"},{"key":"10.1016\/j.humpath.2025.105764_bib23","doi-asserted-by":"crossref","first-page":"594","DOI":"10.5858\/arpa.2015-0170-RS","article-title":"Angiomyolipoma with epithelial cysts","volume":"140","author":"LeRoy","year":"2016","journal-title":"Arch Pathol Lab Med"},{"key":"10.1016\/j.humpath.2025.105764_bib24","first-page":"11760","article-title":"Renal angiomyolipoma with epithelial cysts: a rare entity and review of literature","volume":"8","author":"Wei","year":"2015","journal-title":"Int J Clin Exp Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib25","doi-asserted-by":"crossref","first-page":"1264","DOI":"10.1097\/PAS.0b013e31822539a1","article-title":"Diffuse expression of PAX2 and PAX8 in the cystic epithelium of mixed epithelial stromal tumor, angiomyolipoma with epithelial cysts, and primary renal synovial sarcoma: evidence supporting renal tubular differentiation","volume":"35","author":"Karafin","year":"2011","journal-title":"Am J Surg Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib26","doi-asserted-by":"crossref","DOI":"10.1016\/j.humpath.2024.105693","article-title":"Genetic validation of a TSC2 immunohistochemistry assay in TSC\/mTOR-pathway altered renal tumors","volume":"154","author":"Akbari","year":"2024","journal-title":"Hum Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib27","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1186\/1746-1596-2-11","article-title":"Angiomyolipoma with epithelial cysts (AMLEC): a rare but distinct variant of angiomyolipoma","volume":"2","author":"Armah","year":"2007","journal-title":"Diagn Pathol"},{"key":"10.1016\/j.humpath.2025.105764_bib28","doi-asserted-by":"crossref","first-page":"433","DOI":"10.3389\/fcell.2020.00433","article-title":"Cathepsin K: the action in and beyond bone","volume":"8","author":"Dai","year":"2020","journal-title":"Front Cell Dev Biol"},{"key":"10.1016\/j.humpath.2025.105764_bib29","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1196\/annals.1346.018","article-title":"The regulation of cathepsin K gene expression","volume":"1068","author":"Troen","year":"2006","journal-title":"Ann N Y Acad Sci"}],"container-title":["Human Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0046817725000516?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0046817725000516?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,4,21]],"date-time":"2025-04-21T07:58:04Z","timestamp":1745222284000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0046817725000516"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3]]},"references-count":29,"alternative-id":["S0046817725000516"],"URL":"https:\/\/doi.org\/10.1016\/j.humpath.2025.105764","relation":{},"ISSN":["0046-8177"],"issn-type":[{"value":"0046-8177","type":"print"}],"subject":[],"published":{"date-parts":[[2025,3]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Examination of the histogenesis of epithelial cysts in angiomyolipoma with epithelial cysts (AMLEC) using GPNMB and TSC2 immunostains","name":"articletitle","label":"Article Title"},{"value":"Human Pathology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.humpath.2025.105764","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2025 Elsevier Inc. All rights are reserved, including those for text and data mining, AI training, and similar technologies.","name":"copyright","label":"Copyright"}],"article-number":"105764"}}