{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T10:58:59Z","timestamp":1792839539926,"version":"4.5.0"},"reference-count":43,"publisher":"BMJ","issue":"5","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["J Clin Pathol"],"accepted":{"date-parts":[[2024,4,21]]},"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:sec>\n                  <jats:title>Aim<\/jats:title>\n                  <jats:p>There is some evidence that von Meyenburg complexes (VMCs) can progress to cholangiocarcinoma (CC). This study aimed to evaluate the prevalence of VMCs in CC cases.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>All hepatic resections and explants with intra-hepatic CC (I-CC) and hilar-CC (H-CC) from 1985 to 2020 were studied. Hepatic resections (n=68) for benign lesions or metastatic colonic carcinoma and 15 cases with cirrhosis without any cancer were used as controls.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>A total of 118 cases of CC (88 I-CC, 30\u2009H-CC) were identified. Of these, 61 (52%) patients had no known background liver disease, and 20 (17%) had cirrhosis. Associated liver disorders included metabolic dysfunction-associated steatohepatitis (23), chronic viral hepatitis B or C (13), biliary disease (primary or secondary sclerosing cholangitis) (8), polycystic kidney disease (6), cryptogenic cirrhosis (5) and others miscellaneous disorders (7). VMCs were present in 34 (39%) of 88 I-CC cases and 7 (23%) of 30\u2009H-CC cases. VMCs were present within the tumour (20 cases), outside the cancer (21 cases) or at both locations (10 cases). VMCs with dysplasia\/carcinoma in situ were seen in 19 of 41 (46%) cases with CC and VMCs. In addition, bile duct adenomas were identified in 6 (5%) of CC. 7% of controls showed the presence of VMCs compared with 35% of CC cases (p&lt;0.05).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>VMCs are seen far more frequently in patients with CC than in the control group. The findings support the hypothesis that VMCs could represent a precursor of CC or a marker for a higher risk of developing CC.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1136\/jcp-2024-209572","type":"journal-article","created":{"date-parts":[[2024,5,10]],"date-time":"2024-05-10T15:00:13Z","timestamp":1715353213000},"page":"300-306","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":5,"title":["von Meyenburg complexes are more frequently associated with cholangiocarcinoma"],"prefix":"10.1136","volume":"78","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3845-0798","authenticated-orcid":false,"given":"Dhanpat","family":"Jain","sequence":"first","affiliation":[{"name":"Department of Pathology, Yale School of Medicine, New Haven, Connecticut, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Binny","family":"Khandakar","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine, Donald Barbara Zucker School of Medicine at Hofstra\/Northwell, Manhasset, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pu","family":"Ni","sequence":"additional","affiliation":[{"name":"Department of Pathology, Molecular and Cell-Based Medicine, Icahn School of Medicine at Mount Sinai, New York City, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barton","family":"Kenney","sequence":"additional","affiliation":[{"name":"Department of Pathology, Middlesex Health, Middletown, Connecticut, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lihui","family":"Qin","sequence":"additional","affiliation":[{"name":"Department of Pathology, Weill Cornell Medical College, New York City, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vikram","family":"Deshpande","sequence":"additional","affiliation":[{"name":"Department of Pathology, Harvard Medical School, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Isabel","family":"Fiel","sequence":"additional","affiliation":[{"name":"Department of Pathology, Molecular and Cell-Based Medicine, Icahn School of Medicine at Mount Sinai, New York City, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2024,5,10]]},"reference":[{"key":"2025092505504632000_78.5.300.1","unstructured":"Saul S . Masses of the liver. In: Sternberg SS , Antonioli DA , Carter D , et al , eds. Diagnostic surgical pathology, 2nd ed. New York, NY: Lippincott-Raven, 1994: 1517\u201380."},{"key":"2025092505504632000_78.5.300.2","doi-asserted-by":"crossref","unstructured":"Wittekind C , Henson D , et al . Hepatocellular carcinoma and cholangiocarcinoma. In: Hermanek P , Gospodarowicz MK , eds. Prognostic factors in cancer. Berlin: Springer, 1995: 88\u201393.","DOI":"10.1007\/978-3-642-79395-0_9"},{"key":"2025092505504632000_78.5.300.3","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19850615)55:12<2826::AID-CNCR2820551219>3.0.CO;2-1"},{"key":"2025092505504632000_78.5.300.4","first-page":"194","article-title":"Cholangiocarcinoma arising in caroli\u2019s disease","volume":"105","author":"Phinney","year":"1981","journal-title":"Arch Pathol Lab Med"},{"key":"2025092505504632000_78.5.300.5","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19821001)50:7<1408::AID-CNCR2820500728>3.0.CO;2-T"},{"key":"2025092505504632000_78.5.300.6","first-page":"113","article-title":"Bile duct carcinoma: a late complication of congenital hepatic fibrosis. Case report and review of literature","volume":"73","author":"Scott","year":"1980","journal-title":"Am J Gastroenterol"},{"key":"2025092505504632000_78.5.300.7","doi-asserted-by":"publisher","DOI":"10.1097\/MEG.0b013e32835fb9ee"},{"key":"2025092505504632000_78.5.300.8","first-page":"221","article-title":"Bile-duct hamartomas presenting as multiple focal lesions on hepatic ultrasonography","volume":"87","author":"Sal\u00f3","year":"1992","journal-title":"Am J Gastroenterol"},{"key":"2025092505504632000_78.5.300.9","first-page":"233","article-title":"The hepatic von meyenburg complex: prevalence and association with hepatic and renal cysts among 2843 autopsies [corrected]","volume":"9","author":"Redston","year":"1996","journal-title":"Mod Pathol"},{"key":"2025092505504632000_78.5.300.10","first-page":"93","article-title":"Biliary hamartomas (von meyenburg complexes) in liver needle biopsies","volume":"86","author":"Thommesen","year":"1978","journal-title":"Acta Pathol Microbiol Scand A"},{"key":"2025092505504632000_78.5.300.11","first-page":"141","article-title":"Occurrence of benign hepatocellular tumors in alcoholic men","volume":"94","author":"Karhunen","year":"1986","journal-title":"Acta Pathol Microbiol Immunol Scand A"},{"key":"2025092505504632000_78.5.300.12","doi-asserted-by":"publisher","DOI":"10.12998\/wjcc.v6.i9.296"},{"key":"2025092505504632000_78.5.300.13","doi-asserted-by":"publisher","DOI":"10.1016\/S0046-8177(79)80048-9"},{"key":"2025092505504632000_78.5.300.14","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-199607000-00009"},{"key":"2025092505504632000_78.5.300.15","doi-asserted-by":"publisher","DOI":"10.1007\/BF00192644"},{"key":"2025092505504632000_78.5.300.16","doi-asserted-by":"publisher","DOI":"10.21873\/anticanres.15344"},{"key":"2025092505504632000_78.5.300.17","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-200008000-00011"},{"key":"2025092505504632000_78.5.300.18","doi-asserted-by":"publisher","DOI":"10.1007\/s10620-007-9777-5"},{"key":"2025092505504632000_78.5.300.19","doi-asserted-by":"publisher","DOI":"10.1097\/PAI.0b013e3181b94fd8"},{"key":"2025092505504632000_78.5.300.20","doi-asserted-by":"publisher","DOI":"10.1002\/path.1700960231"},{"key":"2025092505504632000_78.5.300.21","first-page":"285","article-title":"The development of cholangiocarcinoma from multiple bile-duct adenomas. Report of a case and review of the literature","volume":"92","author":"B\u00f6rnfors","year":"1984","journal-title":"Acta Pathol Microbiol Immunol Scand A"},{"key":"2025092505504632000_78.5.300.22","doi-asserted-by":"publisher","DOI":"10.1016\/S0046-8177(86)80575-5"},{"key":"2025092505504632000_78.5.300.23","doi-asserted-by":"publisher","DOI":"10.1007\/BF01540285"},{"key":"2025092505504632000_78.5.300.24","first-page":"1287","article-title":"Cholangiocarcinoma in association with multiple biliary microhamartomas","volume":"114","author":"Burns","year":"1990","journal-title":"Arch Pathol Lab Med"},{"key":"2025092505504632000_78.5.300.25","doi-asserted-by":"publisher","DOI":"10.1016\/0168-9525(93)90209-Z"},{"key":"2025092505504632000_78.5.300.26","first-page":"27","article-title":"Precancerous bile duct pathology in end-stage primary sclerosing cholangitis, with and without cholangiocarcinoma","volume":"34","author":"Lewis","year":"2010","journal-title":"Am J Surg"},{"key":"2025092505504632000_78.5.300.27","doi-asserted-by":"publisher","DOI":"10.1016\/S0168-8278(00)00034-9"},{"key":"2025092505504632000_78.5.300.28","first-page":"346","article-title":"Cholangiocarcinoma arising from biliary micro-hamartomas in a man suffering from hemochromatosis","volume":"17","author":"Yaziji","year":"1997","journal-title":"Ann Pathol"},{"key":"2025092505504632000_78.5.300.29","doi-asserted-by":"publisher","DOI":"10.1097\/00004836-199609000-00018"},{"key":"2025092505504632000_78.5.300.30","doi-asserted-by":"publisher","DOI":"10.1111\/j.1699-0463.1961.tb03202.x"},{"key":"2025092505504632000_78.5.300.31","doi-asserted-by":"publisher","DOI":"10.1097\/MEG.0b013e3282fc73b1"},{"key":"2025092505504632000_78.5.300.32","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2017.08.004"},{"key":"2025092505504632000_78.5.300.33","doi-asserted-by":"publisher","DOI":"10.1097\/PAI.0000000000000132"},{"key":"2025092505504632000_78.5.300.34","doi-asserted-by":"publisher","DOI":"10.1111\/j.1440-1827.2008.02264.x"},{"key":"2025092505504632000_78.5.300.35","first-page":"4109","article-title":"Von-meyenburg complexes are colonic adenoma equivalents in liver","volume":"22","author":"Jain","year":"2009","journal-title":"Mod Pathol"},{"key":"2025092505504632000_78.5.300.36","first-page":"922","article-title":"The bile duct adenoma. A lesion distinct from meyenburg complex","volume":"108","author":"Govindarajan","year":"1984","journal-title":"Arch Pathol Lab Med"},{"key":"2025092505504632000_78.5.300.37","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-198809000-00007"},{"key":"2025092505504632000_78.5.300.38","first-page":"1312","article-title":"An immunohistochemical profile of the so-called bile duct adenoma: clues to pathogenesis","volume":"34","author":"Hughes","year":"2010","journal-title":"Am J Surg"},{"key":"2025092505504632000_78.5.300.39","doi-asserted-by":"publisher","DOI":"10.1097\/00004728-199805000-00006"},{"key":"2025092505504632000_78.5.300.40","doi-asserted-by":"publisher","DOI":"10.1016\/j.anndiagpath.2006.12.003"},{"key":"2025092505504632000_78.5.300.41","doi-asserted-by":"publisher","DOI":"10.1016\/j.anndiagpath.2010.04.003"},{"key":"2025092505504632000_78.5.300.42","doi-asserted-by":"publisher","DOI":"10.1002\/path.1711040305"},{"key":"2025092505504632000_78.5.300.43","doi-asserted-by":"crossref","first-page":"982","DOI":"10.1016\/j.bbadis.2018.03.026","article-title":"Animal models of cholangiocarcinoma","volume":"1865","author":"Loeuillard","year":"2019","journal-title":"Biochim Biophys Acta Mol Basis Dis"}],"container-title":["Journal of Clinical Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/jcp-2024-209572","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,9,25]],"date-time":"2025-09-25T12:50:58Z","timestamp":1758804658000},"score":1,"resource":{"primary":{"URL":"https:\/\/jcp.bmj.com\/lookup\/doi\/10.1136\/jcp-2024-209572"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,10]]},"references-count":43,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2025,4,17]]},"published-print":{"date-parts":[[2025,5]]}},"alternative-id":["10.1136\/jcp-2024-209572"],"URL":"https:\/\/doi.org\/10.1136\/jcp-2024-209572","relation":{},"ISSN":["0021-9746","1472-4146"],"issn-type":[{"value":"0021-9746","type":"print"},{"value":"1472-4146","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,10]]}}}