{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T08:54:06Z","timestamp":1776329646131,"version":"3.50.1"},"reference-count":25,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,3,9]],"date-time":"2026-03-09T00:00:00Z","timestamp":1773014400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"},{"start":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T00:00:00Z","timestamp":1776297600000},"content-version":"vor","delay-in-days":38,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"DOI":"10.1186\/s13256-026-05955-7","type":"journal-article","created":{"date-parts":[[2026,3,9]],"date-time":"2026-03-09T14:36:31Z","timestamp":1773066991000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Localized cystic dilatation of the intrahepatic bile ducts as a distinct subtype of Caroli disease: a case report"],"prefix":"10.1186","volume":"20","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4645-5556","authenticated-orcid":false,"given":"Shoko","family":"Kato","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kenitiro","family":"Kaneko","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuo","family":"Noda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Nouso","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasuyuki","family":"Fukami","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tsuyoshi","family":"Sano","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2026,3,9]]},"reference":[{"key":"5955_CR1","first-page":"488","volume":"34","author":"J Caroli","year":"1958","unstructured":"Caroli J, Soupault R, Kossakowski J, Plocker L. Paradowska. La dilatation polykystique cong\u00e9nitale des voies biliaires intra-h\u00e9patiques; essai de classification [Congenital polycystic dilation of the intrahepatic bile ducts; attempt at classification]. Sem Hop. 1958;34:488\u201395.","journal-title":"Sem Hop"},{"key":"5955_CR2","doi-asserted-by":"publisher","first-page":"340","DOI":"10.1007\/s00534-002-0733-7","volume":"10","author":"T Todani","year":"2003","unstructured":"Todani T, Watanabe Y, Toki A, Morotomi Y. Classification of congenital biliary cystic disease: special reference to type Ic and IVA cysts with primary ductal stricture. J Hepatobiliary Pancreat Surg. 2003;10:340\u20134.","journal-title":"J Hepatobiliary Pancreat Surg"},{"key":"5955_CR3","doi-asserted-by":"publisher","first-page":"713","DOI":"10.1097\/SLA.0000000000000269","volume":"258","author":"JY Mabrut","year":"2013","unstructured":"Mabrut JY, Kianmanesh R, Nuzzo G, Castaing D, Boudjema K, L\u00e9toublon C, et al. Surgical management of congenital intrahepatic bile duct dilatation, Caroli\u2019s disease and syndrome: long-term results of the French Association of Surgery Multicenter Study. Ann Surg. 2013;258:713\u201321.","journal-title":"Ann Surg"},{"key":"5955_CR4","first-page":"167","volume":"128","author":"P Guntz","year":"1991","unstructured":"Guntz P, Coppo B, Lorimier G, Cronier P, Guntz M. La maladie de Caroli unilobaire. Aspects anatomocliniques. D\u00e9marche diagnostique et th\u00e9rapeutique. A propos de 3 observations personnelles et de 101 observations de la litt\u00e9rature [Single-lobe Caroli\u2019s disease. Anatomoclinical aspects. Diagnostic and therapeutic procedure. Apropos of 3 personal cases and 101 cases in the literature]. J Chir (Paris). 1991;128:167\u201381.","journal-title":"J Chir (Paris)"},{"key":"5955_CR5","doi-asserted-by":"publisher","first-page":"711","DOI":"10.1097\/00000658-197110000-00014","volume":"174","author":"WP Longmire Jr","year":"1971","unstructured":"Longmire WP Jr, Mandiola SA, Gordon HE. Congenital cystic disease of the liver and biliary system. Ann Surg. 1971;174:711\u201326.","journal-title":"Ann Surg"},{"key":"5955_CR6","doi-asserted-by":"publisher","first-page":"2019","DOI":"10.1007\/s11605-015-2918-9","volume":"19","author":"MA Moslim","year":"2015","unstructured":"Moslim MA, Gunasekaran G, Vogt D, Cruise M, Morris-Stiff G. Surgical management of Caroli\u2019s disease: single center experience and review of the literature. J Gastrointest Surg. 2015;19:2019\u201327.","journal-title":"J Gastrointest Surg"},{"key":"5955_CR7","doi-asserted-by":"publisher","first-page":"92","DOI":"10.1186\/s40792-021-01175-x","volume":"7","author":"S Kato","year":"2021","unstructured":"Kato S, Kaneko K, Matsushita N, Kurahashi S, Osawa T, Matsumura T, et al. Calcium bilirubinate sludge causes early onset of congenital biliary dilatation: a report of two cases. Surg Case Rep. 2021;7:92.","journal-title":"Surg Case Rep"},{"key":"5955_CR8","doi-asserted-by":"publisher","first-page":"617","DOI":"10.1136\/jcp.50.7.617","volume":"50","author":"K Ohmoto","year":"1997","unstructured":"Ohmoto K, Shimizu M, Iguchi Y, Yamamoto S, Murakami M, Tsunoda T. Solitary cystic dilatation of the intrahepatic bile duct. J Clin Pathol. 1997;50:617\u20138.","journal-title":"J Clin Pathol"},{"key":"5955_CR9","doi-asserted-by":"publisher","first-page":"1001","DOI":"10.1111\/j.1365-2559.1985.tb02884.x","volume":"9","author":"MJ Payan","year":"1985","unstructured":"Payan MJ, Choux R, Sahel J, Laugier R, Kennedy R, Sastre B, et al. Caroli\u2019s disease associated with pancreatic heterotopia and biliary papillomatosis. Histopathology. 1985;9:1001\u20136.","journal-title":"Histopathology"},{"key":"5955_CR10","doi-asserted-by":"publisher","first-page":"271","DOI":"10.1155\/1999\/70985","volume":"11","author":"EK Abdalla","year":"1999","unstructured":"Abdalla EK, Forsmark CE, Lauwers GY, Vauthey JN. Monolobar Caroli\u2019s disease and cholangiocarcinoma. HPB Surg. 1999;11:271\u20136.","journal-title":"HPB Surg"},{"key":"5955_CR11","first-page":"1301","volume":"82","author":"T Terada","year":"1987","unstructured":"Terada T, Nakanuma Y. Solitary cystic dilation of the intrahepatic bile duct: morphology of two autopsy cases and a review of the literature. Am J Gastroenterol. 1987;82:1301\u20135.","journal-title":"Am J Gastroenterol"},{"key":"5955_CR12","unstructured":"Kaneko K, Ishihara S, Ono S, Miyoshi H, Morotomi Y. Atlas of Pancreaticobiliary Maljunction & Congenital Biliary Dilatation. Japanese Study Group on Pancreaticobiliary Maljunction (JSPBM) Scientific Committee. https:\/\/www.jspbm.jp\/en\/."},{"key":"5955_CR13","doi-asserted-by":"publisher","first-page":"1069","DOI":"10.1002\/hep.1840160434","volume":"16","author":"VJ Desmet","year":"1992","unstructured":"Desmet VJ. Congenital diseases of intrahepatic bile ducts: variations on the theme \u201cductal plate malformation.\u201d Hepatology. 1992;16:1069\u201383.","journal-title":"Hepatology"},{"key":"5955_CR14","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1016\/S1665-2681(19)31893-9","volume":"7","author":"JA Calvo-Ponce","year":"2008","unstructured":"Calvo-Ponce JA, Reyes-Richa RV, Rodr\u00edguez Zentner HA. Cyst of the common hepatic duct: treatment and proposal for a modification of Todani\u2019s classification. Ann Hepatol. 2008;7:80\u20132.","journal-title":"Ann Hepatol"},{"key":"5955_CR15","doi-asserted-by":"publisher","first-page":"1059","DOI":"10.3390\/diagnostics13061059","volume":"13","author":"A Miron","year":"2023","unstructured":"Miron A, Popa LG, Toma EA, Calu V, Parvuletu RF, Enciu O. The curious case of the choledochal cyst-revisiting the Todani classification: case report and review of the literature. Diagnostics (Basel). 2023;13:1059.","journal-title":"Diagnostics (Basel)"},{"key":"5955_CR16","doi-asserted-by":"publisher","first-page":"68","DOI":"10.3919\/ringe1963.45.68","volume":"45","author":"T Seno","year":"1984","unstructured":"Seno T, Murase N, Kato K, Akanabe K, Taguchi M, Kinoshita E. A case of intrahepatic choledochal cyst. J Jpn Pract Surg Soc. 1984;45:68\u201373. https:\/\/doi.org\/10.3919\/ringe1963.45.68. ([In Japanese]).","journal-title":"J Jpn Pract Surg Soc"},{"key":"5955_CR17","first-page":"692","volume":"47","author":"A Kuroda","year":"1985","unstructured":"Kuroda A, Kaji T, Nagai H, Morioka Y. Adult intrahepatic biliary dilatation. Surgery. 1985;47:692\u2013701 ([In Japanese]).","journal-title":"Surgery"},{"key":"5955_CR18","doi-asserted-by":"publisher","first-page":"1248","DOI":"10.1016\/0022-3468(90)90520-J","volume":"25","author":"DK Brown","year":"1990","unstructured":"Brown DK, Kimura K, Sato Y, Pringle KC, Abu-Yousef MM, Soper RT. Solitary intrahepatic biliary cyst: diagnostic and therapeutic strategy. J Pediatr Surg. 1990;25:1248\u20139.","journal-title":"J Pediatr Surg"},{"key":"5955_CR19","first-page":"372","volume":"11","author":"M Yamazaki","year":"1997","unstructured":"Yamazaki M, Watanabe Y, Irie U, Ishijima H, Ota M, Sato H. Solitary cystic dilatation of the intrahepatic bile duct \u2013 a case report and a review of the literature. Jpn J Biliary Assoc. 1997;11:372\u20137 ([In Japanese, English abstract]).","journal-title":"Jpn J Biliary Assoc"},{"key":"5955_CR20","doi-asserted-by":"publisher","first-page":"333","DOI":"10.1016\/j.jpedsurg.2006.10.024","volume":"42","author":"TN Rogers","year":"2007","unstructured":"Rogers TN, Woodley H, Ramsden W, Wyatt JI, Stringer MD. Solitary liver cysts in children: not always so simple. J Pediatr Surg. 2007;42:333\u20139.","journal-title":"J Pediatr Surg"},{"key":"5955_CR21","doi-asserted-by":"publisher","first-page":"245","DOI":"10.1007\/s00383-013-3329-1","volume":"30","author":"ZD Burke","year":"2014","unstructured":"Burke ZD, Boechat I, Tabsh K, Deugarte DA. Solitary diverticular type V bile duct cyst. Pediatr Surg Int. 2014;30:245\u20137.","journal-title":"Pediatr Surg Int"},{"key":"5955_CR22","first-page":"788","volume":"15","author":"AD Gidi","year":"2016","unstructured":"Gidi AD, Gonz\u00e1lez-Ch\u00e1vez MA, Villegas-Tovar E, Visag-Castillo V, Pantoja-Millan JP, V\u00e9lez-P\u00e9rez FM, et al. An unusual type of biliar cyst: a case report. Ann Hepatol. 2016;15:788\u201394.","journal-title":"Ann Hepatol"},{"key":"5955_CR23","doi-asserted-by":"publisher","unstructured":"Miyake K, Kawashima H, Ishimaru T, Ihara Y, Hattori K, Oiki H, et al. A case difficult to distinguish between Todani Type II and V. Proceedings of Japan Study Group on Pancreaticobiliary Maljunction. 2021;44:32\u20133. https:\/\/doi.org\/10.34410\/jspbm.44.0_32 [In Japanese]","DOI":"10.34410\/jspbm.44.0_32"},{"key":"5955_CR24","doi-asserted-by":"publisher","first-page":"e945049","DOI":"10.12659\/AJCR.945049","volume":"25","author":"TH Alghamdi","year":"2024","unstructured":"Alghamdi TH. Intrahepatic Class VII choledochal cyst: radiological insights and surgical approach. Am J Case Rep. 2024;25:e945049.","journal-title":"Am J Case Rep"},{"key":"5955_CR25","doi-asserted-by":"publisher","first-page":"267","DOI":"10.1016\/j.hpb.2021.06.423","volume":"24","author":"MH Fard-Aghaie","year":"2022","unstructured":"Fard-Aghaie MH, Makridis G, Reese T, Feyerabend B, Wagner KC, Schnitzbauer A, et al. The rate of cholangiocarcinoma in Caroli disease. A German multicenter study. HPB (Oxford). 2022;24:267\u201376.","journal-title":"HPB (Oxford)"}],"container-title":["Journal of Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13256-026-05955-7","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-026-05955-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-026-05955-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T07:52:35Z","timestamp":1776325955000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s13256-026-05955-7"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3,9]]},"references-count":25,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["5955"],"URL":"https:\/\/doi.org\/10.1186\/s13256-026-05955-7","relation":{},"ISSN":["1752-1947"],"issn-type":[{"value":"1752-1947","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3,9]]},"assertion":[{"value":"1 May 2025","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"30 January 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 March 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The ethics committee of Aichi Medical University approved this case report. Informed consent was obtained from the patients\u2019 legal guardians.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patients\u2019 legal guardians for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"199"}}