{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,7]],"date-time":"2026-09-07T08:09:22Z","timestamp":1788768562708,"version":"build-2803163510"},"reference-count":17,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2026,9,7]],"date-time":"2026-09-07T00:00:00Z","timestamp":1788739200000},"content-version":"vor","delay-in-days":7,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,8,31]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Caroli disease is characterized by segmental, non-obstructive dilatation of the intrahepatic bile ducts, leading to bile stasis, recurrent cholangitis, hepatolithiasis, and an increased risk of cholangiocarcinoma. Caroli\u2019s disease, together with hepatic fibrosis, is termed Caroli\u2019s syndrome, which may lead to portal hypertension. Caroli\u2019s disease often requires treatment in symptomatic patients. Symptoms include recurrent cholangitis due to intrahepatic stones in dilated ducts or cirrhosis due to fibrosis. Liver transplantation remains the definitive treatment for diffuse disease. However, owing to organ shortages and long waiting lists, liver resection is an alternative for localized disease. Here, we present a rare case of an 18-year-old male with symptomatic, right-lobe-predominant Caroli\u2019s disease and hepatolithiasis who underwent surgical resection (right hepatectomy, stone extraction from exposed ducts at the resection margin, and intrahepatic-jejunostomy) with symptom resolution. This case highlights the role of liver resection in localized disease and reinforces the importance of individualized interventions in resource-limited settings.<\/jats:p>","DOI":"10.1093\/jscr\/rjag769","type":"journal-article","created":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T11:37:58Z","timestamp":1788521878000},"source":"Crossref","is-referenced-by-count":0,"title":["Diffuse Caroli\u2019s disease with hepatolithiasis: a case report"],"prefix":"10.1093","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-9647-8240","authenticated-orcid":false,"given":"Shruti","family":"Sah","sequence":"first","affiliation":[{"name":"Department of Gastrointestinal Surgery, Birat Medical College Teaching Hospital , Tankisinuwari, Morang, Biratnagar 56613, Province 1 ,","place":["Nepal"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-7741-5705","authenticated-orcid":false,"given":"Ranjish","family":"Parshaila","sequence":"additional","affiliation":[{"name":"Department of Gastrointestinal Surgery, Birat Medical College Teaching Hospital , Tankisinuwari, Morang, Biratnagar 56613, Province 1 ,","place":["Nepal"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dinesh","family":"Nalbo","sequence":"additional","affiliation":[{"name":"Department of Gastrointestinal Surgery, Birat Medical College Teaching Hospital , Tankisinuwari, Morang, Biratnagar 56613, Province 1 ,","place":["Nepal"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6904-7972","authenticated-orcid":false,"given":"Narendra","family":"Pandit","sequence":"additional","affiliation":[{"name":"Department of Gastrointestinal Surgery, Birat Medical College Teaching Hospital , Tankisinuwari, Morang, Biratnagar 56613, Province 1 ,","place":["Nepal"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,9,7]]},"reference":[{"key":"2026090703371620500_ref1","doi-asserted-by":"publisher","first-page":"578","DOI":"10.1097\/MEG.0b013e3283470fcd","article-title":"Caroli disease: review of eight cases with emphasis on magnetic resonance imaging features","volume":"23","author":"Lefere","year":"2011","journal-title":"Eur J Gastroenterol Hepatol"},{"key":"2026090703371620500_ref2","volume-title":"StatPearls [Internet]","author":"Kyalwazi","year":"2025"},{"key":"2026090703371620500_ref3","first-page":"603","article-title":"Sj\u00fakd\u00f3mur Carolis--sj\u00fakratilfelli og yfirlit fraedigreina [Caroli's disease, case report and review of the literature]","volume":"93","author":"Agustsson","year":"2007","journal-title":"Laeknabladid"},{"key":"2026090703371620500_ref4","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1016\/s1479-666x(06)80039-9","article-title":"Partial hepatectomy is curative for the localized type of Caroli's disease: a case report and review of the literature","volume":"4","author":"Yilmaz","year":"2006","journal-title":"Surgeon"},{"key":"2026090703371620500_ref5","doi-asserted-by":"publisher","first-page":"e50871","DOI":"10.7759\/cureus.50871","article-title":"Caroli's syndrome: a case report and literature review","volume":"15","author":"Shafqat","year":"2023","journal-title":"Cureus"},{"key":"2026090703371620500_ref6","doi-asserted-by":"publisher","first-page":"659","DOI":"10.1148\/rg.253045114","article-title":"Fibropolycystic liver disease: CT and MR imaging findings","volume":"25","author":"Brancatelli","year":"2005","journal-title":"Radiographics"},{"key":"2026090703371620500_ref7","doi-asserted-by":"publisher","first-page":"1069","DOI":"10.1002\/hep.1840160434","article-title":"Congenital diseases of intrahepatic bile ducts: variations on the theme \u201cductal plate malformation\u201d","volume":"16","author":"Desmet","year":"1992","journal-title":"Hepatology"},{"key":"2026090703371620500_ref8","doi-asserted-by":"publisher","first-page":"e9135","DOI":"10.7759\/cureus.9135","article-title":"Caroli disease: a presentation of acute pancreatitis and cholangitis","volume":"12","author":"Khan","year":"2020","journal-title":"Cureus"},{"key":"2026090703371620500_ref9","doi-asserted-by":"publisher","first-page":"e1701","DOI":"10.7759\/cureus.1701","article-title":"Caroli's disease as a cause of chronic epigastric abdominal pain: two case reports and a brief review of the literature","volume":"9","author":"Cabral Correia","year":"2017","journal-title":"Cureus"},{"key":"2026090703371620500_ref10","doi-asserted-by":"publisher","first-page":"11","DOI":"10.1002\/lt.21366","article-title":"Caroli's disease and outcomes after liver transplantation","volume":"14","author":"Millwala","year":"2008","journal-title":"Liver Transpl"},{"key":"2026090703371620500_ref11","first-page":"194","article-title":"Cholangiocarcinoma arising in Caroli's disease","volume":"105","author":"Phinney","year":"1981","journal-title":"Arch Pathol Lab Med"},{"key":"2026090703371620500_ref12","doi-asserted-by":"publisher","first-page":"718","DOI":"10.1097\/00000658-198412000-00008","article-title":"Successful treatment of Caroli's disease by hepatic resection","volume":"200","author":"Nagasue","year":"1984","journal-title":"Rep Six Patients Ann Surg"},{"key":"2026090703371620500_ref13","doi-asserted-by":"publisher","first-page":"888","DOI":"10.1016\/j.surg.2005.05.002","article-title":"Caroli's disease: liver resection and liver 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Experience in 33 patients","volume":"138","author":"Kassahun","year":"2005","journal-title":"Surgery"},{"key":"2026090703371620500_ref14","first-page":"189","article-title":"Liver transplantation for caroli disease","volume":"3","author":"Zahmatkeshan","year":"2012","journal-title":"Int J Organ Transplant Med"},{"key":"2026090703371620500_ref15","doi-asserted-by":"publisher","first-page":"1934","DOI":"10.3748\/wjg.v13.i13.1934","article-title":"Clinical characteristics of Caroli's syndrome","volume":"13","author":"Yonem","year":"2007","journal-title":"World J Gastroenterol"},{"key":"2026090703371620500_ref16","doi-asserted-by":"publisher","first-page":"4718","DOI":"10.3748\/wjg.v26.i31.4718","article-title":"Risk of malignancy in Caroli disease and syndrome: a systematic review","volume":"26","author":"Fahrner","year":"2020","journal-title":"World J Gastroenterol"},{"key":"2026090703371620500_ref17","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1002\/bjs.5531","article-title":"Intrahepatic cholangiojejunostomy following hepatobiliary resection","volume":"94","author":"Nagino","year":"2007","journal-title":"Br J Surg"}],"container-title":["Journal of Surgical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jscr\/article-pdf\/2026\/9\/rjag769\/70971822\/rjag769.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jscr\/article-pdf\/2026\/9\/rjag769\/70971822\/rjag769.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,7]],"date-time":"2026-09-07T07:37:44Z","timestamp":1788766664000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jscr\/article\/doi\/10.1093\/jscr\/rjag769\/8786374"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,31]]},"references-count":17,"journal-issue":{"issue":"9","published-print":{"date-parts":[[2026,8,31]]}},"URL":"https:\/\/doi.org\/10.1093\/jscr\/rjag769","relation":{},"ISSN":["2042-8812"],"issn-type":[{"value":"2042-8812","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026,9]]},"published":{"date-parts":[[2026,8,31]]},"article-number":"rjag769"}}