{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T12:30:08Z","timestamp":1790771408123,"version":"4.1.0"},"reference-count":23,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2020,8,3]],"date-time":"2020-08-03T00:00:00Z","timestamp":1596412800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Liver International"],"published-print":{"date-parts":[[2020,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background and Aims<\/jats:title><jats:p>Patients with a choledochal malformation, formerly described as cysts, are at increased risk of developing a cholangiocarcinoma and resection is recommended. Given the low incidence of choledochal malformation (CM) in Western countries, the incidence in these countries is unclear. Our aim was to assess the incidence of malignancy in CM patients and to assess postoperative outcome.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>In a nationwide, retrospective study, all adult patients who underwent surgery for CM between 1990 and 2016 were included. Patients were identified through the Dutch Pathology Registry and local patient records and were analysed to determine the incidence of malignancy, as well as postoperative mortality and morbidity.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 123 patients with a CM were included in the study (Todani Type I, n\u00a0=\u00a071; Type II, n\u00a0=\u00a010; Type III, n\u00a0=\u00a03; Type IV, n\u00a0=\u00a027; unknown, n\u00a0=\u00a012). Median age was 40\u00a0years (range 18\u201070) and 81% were female. The majority of patients (99\/123) underwent extrahepatic bile duct resection, with additional liver parenchyma resections in eight patients, only exploration in two, and a local cyst resection in eight patients. Postoperative 30\u2010day mortality was 2% (2\/123) and limited to patients who underwent liver resection. Severe morbidity occurred in 24%. In 14 of the 123 patients (11%), a malignancy was found in the resected specimen. One patient developed a periampullary malignancy 7\u00a0years later.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>In a large Western series of CM patients, 11% were found to have a malignancy. This justifies resection in these patients, despite the risk of morbidity (24%) and mortality (2%).<\/jats:p><\/jats:sec>","DOI":"10.1111\/liv.14568","type":"journal-article","created":{"date-parts":[[2020,6,20]],"date-time":"2020-06-20T05:35:39Z","timestamp":1592631339000},"page":"2469-2475","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Choledochal malformations in adults in the Netherlands: Results from a nationwide retrospective cohort study"],"prefix":"10.1111","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3975-3184","authenticated-orcid":false,"given":"Ruben H.","family":"de Kleine","sequence":"first","affiliation":[{"name":"Division of Hepato\u2010Pancreato\u2010Biliary Surgery and Liver Transplantation Department of Surgery University of Groningen University Medical Center Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9942-6922","authenticated-orcid":false,"given":"A. Marthe","family":"Schreuder","sequence":"additional","affiliation":[{"name":"Department of Surgery Cancer Center Amsterdam Amsterdam UMC University of Amsterdam Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anneke","family":"ten Hove","sequence":"additional","affiliation":[{"name":"Division of Hepato\u2010Pancreato\u2010Biliary Surgery and Liver Transplantation Department of Surgery University of Groningen University Medical Center Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan B. F.","family":"Hulscher","sequence":"additional","affiliation":[{"name":"Division of Pediatric surgery Department of Surgery University of Groningen University Medical Center Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Inne H. M.","family":"Borel Rinkes","sequence":"additional","affiliation":[{"name":"Department of Surgery University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cornelis H. C.","family":"Dejong","sequence":"additional","affiliation":[{"name":"Department of Surgery University Medical Center Maastricht Maastricht the Netherlands"},{"name":"Department of Surgery RWTH Uniklinikum Aachen Aachen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeroen","family":"de Jonge","sequence":"additional","affiliation":[{"name":"Department of Surgery Erasmus Medical Center Rotterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philip","family":"de Reuver","sequence":"additional","affiliation":[{"name":"Department of Surgery Radboud University Nijmegen Medical Center Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joris","family":"Erdmann","sequence":"additional","affiliation":[{"name":"Department of Surgery Cancer Center Amsterdam Amsterdam UMC University of Amsterdam Amsterdam the Netherlands"},{"name":"Department of Surgery University of Leiden Leiden University Medical Center Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Geert","family":"Kazemier","sequence":"additional","affiliation":[{"name":"Department of Surgery Cancer Center Amsterdam Amsterdam UMC Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"the PALGA Foundation","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas M.","family":"van Gulik","sequence":"additional","affiliation":[{"name":"Department of Surgery Cancer Center Amsterdam Amsterdam UMC University of Amsterdam Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annette S. H.","family":"Gouw","sequence":"additional","affiliation":[{"name":"Department of Pathology University of Groningen University Medical Center Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert J.","family":"Porte","sequence":"additional","affiliation":[{"name":"Division of Hepato\u2010Pancreato\u2010Biliary Surgery and Liver Transplantation Department of Surgery University of Groningen University Medical Center Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,8,3]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/bjs.10798"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/bjs.4815"},{"issue":"5","key":"e_1_2_7_4_1","first-page":"434","article-title":"Choledochal cysts: part 1 of 3: classification and pathogenesis","volume":"52","author":"Singham J","year":"2009","journal-title":"Can J Surg"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00268-016-3684-x"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00268-017-4064-x"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2017.03.003"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0034-1387947"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/jhbp.346"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2014.06.016"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/SLA.0000000000000269"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamasurg.2015.0226"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.0040297"},{"issue":"1","key":"e_1_2_7_14_1","first-page":"19","article-title":"Pathology databanking and biobanking in The Netherlands, a central role for PALGA, the nationwide histopathology and cytopathology data network and archive","volume":"29","author":"Casparie M","year":"2007","journal-title":"Cell Oncol"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9610(77)90359-2"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.sla.0000133083.54934.ae"},{"key":"e_1_2_7_17_1","unstructured":"(CBS) CBvdS.Kerncijfers bevolking.2000.https:\/\/opendata.cbs.nl\/statline"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gassur.2003.12.013"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.surg.2019.01.010"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1001\/archsurg.2011.243"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00268-011-1009-7"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11605-016-3181-4"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jamcollsurg.2006.07.048"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1007\/s005340050108"}],"container-title":["Liver International"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fliv.14568","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/liv.14568","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/liv.14568","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/liv.14568","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,3]],"date-time":"2023-09-03T15:07:39Z","timestamp":1693753659000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/liv.14568"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,8,3]]},"references-count":23,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2020,10]]}},"alternative-id":["10.1111\/liv.14568"],"URL":"https:\/\/doi.org\/10.1111\/liv.14568","archive":["Portico"],"relation":{},"ISSN":["1478-3223","1478-3231"],"issn-type":[{"value":"1478-3223","type":"print"},{"value":"1478-3231","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,8,3]]},"assertion":[{"value":"2020-02-18","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-06-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-08-03","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}