{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T06:40:01Z","timestamp":1756363201042,"version":"3.44.0"},"reference-count":11,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2014,6,9]]},"abstract":"<jats:p>A 31-year-old man presented with features of recurrent cholangitis for 7\u2005months. Examination revealed icterus and a palpable 10\u00d78\u2005cm lump in the right hypochondrium extending into the epigastrium. Total and direct bilirubin was raised (8.4, 6.7\u2005mg\/dL). Alkaline phosphatase (468\u2005U\/L) was raised but other liver enzymes were normal. Ultrasound of the abdomen showed dilated intrahepatic biliary radicals (IHBRs) and common hepatic duct (CHD) with a significant intrahepatic portion. Contrast-enhanced CT scan of the abdomen showed similar findings. MR cholangiopancreatography demonstrated diffusely dilated IHBRs and choledochal cyst involving the CHD. The common bile duct was mildly dilated without any filling defect. Tumour markers (carcinoembryonic antigen and cancer antigen 19-9) were normal. The patient underwent complete excision of the extrahepatic biliary system including choledochal cyst and Roux-en-Y hepaticojejunostomy. Opening up the bile duct showed very thick gelatinous material and multiple pedunculated papillary type structures arising from the wall of the choledochal cyst.<\/jats:p>","DOI":"10.1136\/bcr-2014-204680","type":"journal-article","created":{"date-parts":[[2014,7,2]],"date-time":"2014-07-02T07:27:23Z","timestamp":1404286043000},"page":"bcr2014204680","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Rare event of biliary papillomatosis arising in a choledochal cyst"],"prefix":"10.1136","volume":"2014","author":[{"given":"Kumar","family":"Nishant","sequence":"first","affiliation":[{"name":"Department of Surgery, Sikkim Manipal Institute of Medical Sciences, Gangtok, Sikkim, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1387-9475","authenticated-orcid":false,"given":"Varun Kumar","family":"Singh","sequence":"additional","affiliation":[{"name":"Department of Surgery, Sikkim Manipal Institute of Medical Sciences, Gangtok, Sikkim, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Barun Kumar","family":"Sharma","sequence":"additional","affiliation":[{"name":"Department of Radiodiagnosis, Sikkim Manipal Institute of Medical Sciences, Gangtok, Sikkim, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2014,7,2]]},"reference":[{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.1","first-page":"982","article-title":"Choledochal cyst and neoplasm: a comprehensive review of 106 cases and presentation of two original cases","volume":"63","author":"Feiber","year":"1997","journal-title":"Am Surg"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.2","doi-asserted-by":"publisher","DOI":"10.1007\/s005950200206"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.3","first-page":"222","article-title":"Biliary papillomatosis with adenocarcinoma arising in choledochal cyst","volume":"30","author":"Negi","year":"2009","journal-title":"Trop Gastroenterol"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.4","doi-asserted-by":"crossref","first-page":"1209","DOI":"10.1016\/0016-5085(93)90969-J","article-title":"Biliary papillomatosis with the point mutation of K-ras gene arising in congenital choledochal cyst","volume":"105","author":"Ohita","year":"1993","journal-title":"Gastroenterology"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.5","doi-asserted-by":"crossref","first-page":"748","DOI":"10.1016\/0016-5085(86)91133-9","article-title":"Obstructive cholangitis secondary to mucus secreted by a solitary papillary bile duct tumor","volume":"90","author":"Styne","year":"1986","journal-title":"Gastroenterology"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.6","first-page":"2253","article-title":"Papillomatosis of the bile ducts: papilloma-carcinoma sequence","volume":"89","author":"Holtkamp","year":"1994","journal-title":"Am J Gastroenterol"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.7","doi-asserted-by":"crossref","first-page":"779","DOI":"10.1016\/S0016-5085(76)80275-2","article-title":"Adenocarcinoma in biliary papillomatosis","volume":"70","author":"Neumann","year":"1976","journal-title":"Gastroenterology"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.8","doi-asserted-by":"publisher","DOI":"10.1186\/1477-7819-4-71"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.9","doi-asserted-by":"publisher","DOI":"10.1046\/j.1440-1673.2000.00803.x"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.10","doi-asserted-by":"publisher","DOI":"10.1002\/bjs.1800831218"},{"key":"2025082723262712000_2014.jul02_1.bcr2014204680.11","first-page":"448","article-title":"Malignant papillomatosis of the bile ducts: a new indication for liver plantation","volume":"84","author":"Rambaud","year":"1989","journal-title":"Am J Gastroenterol"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2014-204680","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T06:26:36Z","timestamp":1756362396000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2014-204680"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,7,2]]},"references-count":11,"alternative-id":["10.1136\/bcr-2014-204680"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2014-204680","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2014,7,2]]}}}