{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T04:22:59Z","timestamp":1772166179120,"version":"3.50.1"},"reference-count":22,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,11,9]],"date-time":"2020-11-09T00:00:00Z","timestamp":1604880000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2020,11,9]],"date-time":"2020-11-09T00:00:00Z","timestamp":1604880000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["World J Surg Onc"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Pancreaticobiliary maljunction (PBM) is a congenital abnormality in which the pancreatic and biliary ducts join anatomically outside the duodenal wall resulting in the regurgitation of pancreatic juice into the biliary tract (pancreatobiliary reflux). Persistent pancreatobiliary reflux causes injury to the epithelium of the biliary tract and promotes the risk of biliary cancer. Intracholecyctic papillary neoplasm (ICPN) has been highlighted in the context of a cholecystic counterpart of intraductal papillary mucinous neoplasm of the pancreas and the bile duct, but the tumorigenesis of ICPNs remains unclear.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>\n                      A 52-year-old Japanese woman was referred for the assessment of dilation of the bile duct. Computed tomography which revealed an enhanced mass in the gallbladder and endoscopic retrograde cholangiopancreatography confirmed that the confluence of the main pancreatic duct and extrahepatic bile duct (EHBD) was located outside the duodenal wall. Under the diagnosis of gallbladder cancer with PBM, cholecystectomy with full thickness dissection, EHBD resection, lymph node dissection, and hepaticojejunostomy were performed. Macroscopic examination of the resected specimen showed that the cystic duct was dilated and joined into the EHBD just above its confluence with the pancreatic duct, and the inflamed change of non-tumorous mucosa of gallbladder indicating that there was considerable mucosal injury due to pancreatobiliary reflux to the gallbladder. Histopathological examination revealed that the gallbladder tumor was a gastric-type ICPN with non-invasive component. Either\n                      <jats:italic>KRAS<\/jats:italic>\n                      gene mutation or p53 protein expression that were known to be associated with the carcinogenesis of biliary cancer under the condition of pancreatobiliary reflux was not detected in the tumor cells of ICPN.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>The present case might suggest that there was no association between PBM and ICPN. To reveal the tumorigenesis of ICPN and its attribution to pancreatobiliary reflux, however, further study is warranted.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12957-020-02072-7","type":"journal-article","created":{"date-parts":[[2020,11,9]],"date-time":"2020-11-09T11:07:07Z","timestamp":1604920027000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Intracholecystic papillary neoplasm arising in a patient with pancreaticobiliary maljunction: a case report"],"prefix":"10.1186","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5682-5480","authenticated-orcid":false,"given":"Toshimitsu","family":"Iwasaki","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasuhiro","family":"Otsuka","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoichi","family":"Miyata","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takahiro","family":"Einama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hironori","family":"Tsujimoto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hideki","family":"Ueno","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sho","family":"Ogata","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoji","family":"Kishi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,11,9]]},"reference":[{"key":"2072_CR1","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1007\/BF02391070","volume":"1","author":"The Japanese Study Group on Pancreaticobiliary Maljunction","year":"1994","unstructured":"The Japanese Study Group on Pancreaticobiliary Maljunction. Diagnostic criteria of pancreaticobiliary maljunction. J Hepato-Biliary-Pancreat Surg. 1994;1:219\u201321.","journal-title":"J Hepato-Biliary-Pancreat Surg"},{"issue":"7","key":"2072_CR2","doi-asserted-by":"publisher","first-page":"731","DOI":"10.1007\/s00535-012-0611-2","volume":"47","author":"T Kamisawa","year":"2012","unstructured":"Kamisawa T, Ando H, Suyama M, Shimada M, Morine Y, Shimada H, et al. Japanese clinical practice guidelines for pancreaticobiliary maljunction. J Gastroenterol. 2012;47(7):731\u201359.","journal-title":"J Gastroenterol"},{"issue":"1","key":"2072_CR3","doi-asserted-by":"publisher","first-page":"45","DOI":"10.1007\/s005340200004","volume":"9","author":"Y Matsumoto","year":"2002","unstructured":"Matsumoto Y, Fujii H, Itakura J, Matsuda M, Nobukawa B, Suda K. Recent advances in pancreaticobiliary maljunction. J Hepato-Biliary-Pancreat Surg. 2002;9(1):45\u201354.","journal-title":"J Hepato-Biliary-Pancreat Surg"},{"issue":"2","key":"2072_CR4","doi-asserted-by":"publisher","first-page":"158","DOI":"10.1007\/s00535-016-1268-z","volume":"52","author":"T Kamisawa","year":"2017","unstructured":"Kamisawa T, Kuruma S, Chiba K, Tabata T, Koizumi S, Kikuyama M. Biliary carcinogenesis in pancreaticobiliary maljunction. J Gastroenterol. 2017;52(2):158\u201363.","journal-title":"J Gastroenterol"},{"key":"2072_CR5","doi-asserted-by":"publisher","first-page":"S84","DOI":"10.1016\/j.cgh.2009.08.024","volume":"7","author":"T Kamisawa","year":"2009","unstructured":"Kamisawa T, Takuma K, Ajiki H, Egawa N, Kurata M, Honda G, et al. Pancreaticobiliary maljunction. Clin Gastroenterol Hepatol. 2009;7:S84\u20138.","journal-title":"Clin Gastroenterol Hepatol"},{"issue":"9","key":"2072_CR6","doi-asserted-by":"publisher","first-page":"1279","DOI":"10.1097\/PAS.0b013e318262787c","volume":"36","author":"V Adsay","year":"2012","unstructured":"Adsay V, Jang KT, Roa JC, Dursun N, Ohike N, Bagci P, et al. Intracholecystic papillary-tubular neoplasms (ICPN) of the gallbladder (neoplastic polyps, adenomas, and papillary neoplasms that are \u22651.0\u2009cm): clinicopathologic and immunohistochemical analysis of 123 cases. Am J Surg Pathol. 2012;36(9):1279\u2013301.","journal-title":"Am J Surg Pathol"},{"key":"2072_CR7","first-page":"266","volume-title":"World Health Organization of tumours","author":"J Albores-Saavedra","year":"2010","unstructured":"Albores-Saavedra J, Adsay N, Crawford J, Kimstra D, Kloppel G, Sripa B. WHO classification of tumours of the digestive system. In: World Health Organization of tumours. 4th ed. Lyon: IARC; 2010. p. 266\u201373.","edition":"4"},{"issue":"6","key":"2072_CR8","doi-asserted-by":"publisher","first-page":"783","DOI":"10.1097\/PAS.0000000000001237","volume":"43","author":"M Akita","year":"2019","unstructured":"Akita M, Fujikura K, Ajiki T, Fukumoto T, Otani K, Hirose T, et al. Intracholecystic papillary neoplasms are distinct from papillary gallbladder cancers: a clinicopathologic and exome-sequencing study. Am J Surg Pathol. 2019;43(6):783\u201391.","journal-title":"Am J Surg Pathol"},{"key":"2072_CR9","doi-asserted-by":"publisher","first-page":"276","DOI":"10.1007\/BF02489025","volume":"4","author":"T Takuji","year":"1997","unstructured":"Takuji T. Congenital choledochal dilation: classification, clinical features, and long-term results. J Hepato-Biliary-Pancreat Surg. 1997;4:276\u201382.","journal-title":"J Hepato-Biliary-Pancreat Surg"},{"issue":"3","key":"2072_CR10","first-page":"522","volume":"60","author":"E Hidaka","year":"2000","unstructured":"Hidaka E, Yanagisawa A, Seki M, Takano K, Setoguchi T, Kato Y. High frequency of K-ras mutations in biliary duct carcinomas of cases with a long common channel in the papilla of Vater. Cancer Res. 2000;60(3):522\u20134.","journal-title":"Cancer Res"},{"issue":"1","key":"2072_CR11","doi-asserted-by":"publisher","first-page":"56","DOI":"10.1016\/S0016-5107(04)01290-8","volume":"60","author":"T Kamisawa","year":"2004","unstructured":"Kamisawa T, Funata N, Hayashi Y, Egawa N, Nakajima H, Tsuruta K, et al. Pathologic changes in the non-carcinomatous epithelium of the gallbladder in patients with a relatively long common channel. Gastrointest Endosc. 2004;60(1):56\u201360.","journal-title":"Gastrointest Endosc"},{"issue":"3","key":"2072_CR12","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1007\/BF02784932","volume":"14","author":"T Tabata","year":"1993","unstructured":"Tabata T, Fujimori T, Maeda S, Yamamoto M, Saitoh Y. The role of Ras mutation in pancreatic cancer, precancerous lesions, and chronic pancreatitis. Int J Pancreatol. 1993;14(3):237\u201344.","journal-title":"Int J Pancreatol"},{"issue":"3","key":"2072_CR13","doi-asserted-by":"publisher","first-page":"130","DOI":"10.4251\/wjgo.v2.i3.130","volume":"2","author":"A Tsuchida","year":"2010","unstructured":"Tsuchida A, Itoi T. Carcinogenesis and chemoprevention of biliary tract cancer in pancreaticobiliary maljunction. World J Gastrointest Oncol. 2010;2(3):130\u20135.","journal-title":"World J Gastrointest Oncol"},{"issue":"3","key":"2072_CR14","doi-asserted-by":"publisher","first-page":"183","DOI":"10.1016\/j.pan.2012.04.004","volume":"12","author":"M Tanaka","year":"2012","unstructured":"Tanaka M, Fernandez-del Castillo C, Adsay V, Chari S, Falconi M, Jang JY, et al. International consensus guidelines 2012 for the management of IPMN and MCN of the pancreas. Pancreatology. 2012;12(3):183\u201397.","journal-title":"Pancreatology."},{"issue":"4 Pt 1","key":"2072_CR15","doi-asserted-by":"publisher","first-page":"651","DOI":"10.1053\/jhep.2001.28199","volume":"34","author":"TC Chen","year":"2001","unstructured":"Chen TC, Nakanuma Y, Zen Y, Chen MF, Jan YY, Yeh TS, et al. Intraductal papillary neoplasia of the liver associated with hepatolithiasis. Hepatology. 2001;34(4 Pt 1):651\u20138.","journal-title":"Hepatology."},{"issue":"12","key":"2072_CR16","doi-asserted-by":"publisher","first-page":"1862","DOI":"10.1097\/PAS.0b013e3182986bb5","volume":"37","author":"JH Tsai","year":"2013","unstructured":"Tsai JH, Yuan RH, Chen YL, Liau JY, Jeng YM. GNAS Is frequently mutated in a specific subgroup of intraductal papillary neoplasms of the bile duct. Am J Surg Pathol. 2013;37(12):1862\u201370.","journal-title":"Am J Surg Pathol"},{"issue":"7","key":"2072_CR17","doi-asserted-by":"publisher","first-page":"1535","DOI":"10.1007\/s00261-018-1595-z","volume":"43","author":"N Mizobuchi","year":"2018","unstructured":"Mizobuchi N, Munechika J, Takeyama N, Ohgiya Y, Ohike N, Abe R, et al. Three cases of intracystic papillary neoplasm of gallbladder. Abdom Radiol. 2018;43(7):1535\u20139.","journal-title":"Abdom Radiol"},{"issue":"5","key":"2072_CR18","doi-asserted-by":"publisher","first-page":"472","DOI":"10.1007\/s00534-013-0606-2","volume":"20","author":"Y Morine","year":"2013","unstructured":"Morine Y, Shimada M, Takamatsu H, Araida T, Endo I, Kubota M, et al. Clinical features of pancreaticobiliary maljunction: update analysis of 2nd Japan-nationwide survey. J Hepatobiliary Pancreat Sci. 2013;20(5):472\u201380.","journal-title":"J Hepatobiliary Pancreat Sci"},{"issue":"12","key":"2072_CR19","doi-asserted-by":"publisher","first-page":"850","DOI":"10.1093\/jjco\/hyp106","volume":"39","author":"T Matsuda","year":"2009","unstructured":"Matsuda T, Marugame T, Kamo K, Katanoda K, Ajiki W, Sobue T, et al. Cancer incidence and incidence rates in Japan in 2003: based on data from 13 population-based cancer registries in the Monitoring of Cancer Incidence in Japan (MCIJ) Project. Jpn J Clin Oncol. 2009;39(12):850\u20138.","journal-title":"Jpn J Clin Oncol"},{"issue":"3","key":"2072_CR20","doi-asserted-by":"publisher","first-page":"376","DOI":"10.1007\/s00534-008-0030-1","volume":"16","author":"K Kasuya","year":"2009","unstructured":"Kasuya K, Nagakawa Y, Matsudo T, Ozawa T, Tsuchida A, Aoki T, et al. p53 gene mutation and p53 protein overexpression in a patient with simultaneous double cancer of the gallbladder and bile duct associated with pancreaticobiliary maljunction. J Hepato-Biliary-Pancreat Surg. 2009;16(3):376\u201381.","journal-title":"J Hepato-Biliary-Pancreat Surg"},{"key":"2072_CR21","doi-asserted-by":"publisher","first-page":"43","DOI":"10.1186\/1742-4682-3-43","volume":"3","author":"RA Ruggiero","year":"2006","unstructured":"Ruggiero RA, Bustuoabad OD. The biological sense of cancer: a hypothesis. Theor Biol Med Model. 2006;3:43.","journal-title":"Theor Biol Med Model"},{"issue":"9","key":"2072_CR22","doi-asserted-by":"publisher","first-page":"465","DOI":"10.1111\/pin.12188","volume":"64","author":"Y Meguro","year":"2014","unstructured":"Meguro Y, Fukushima N, Koizumi M, Kasahara N, Hydo M, Morishima K, et al. A case of mixed adenoneuroendocrine carcinoma of the gallbladder arising from an intracystic papillary neoplasm associated with pancreaticobiliary maljunction. Pathol Int. 2014;64(9):465\u201371.","journal-title":"Pathol Int"}],"container-title":["World Journal of Surgical Oncology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s12957-020-02072-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s12957-020-02072-7\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s12957-020-02072-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,11,27]],"date-time":"2022-11-27T05:50:01Z","timestamp":1669528201000},"score":1,"resource":{"primary":{"URL":"https:\/\/wjso.biomedcentral.com\/articles\/10.1186\/s12957-020-02072-7"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,11,9]]},"references-count":22,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["2072"],"URL":"https:\/\/doi.org\/10.1186\/s12957-020-02072-7","relation":{"has-preprint":[{"id-type":"doi","id":"10.21203\/rs.3.rs-61716\/v2","asserted-by":"object"},{"id-type":"doi","id":"10.21203\/rs.3.rs-61716\/v1","asserted-by":"object"}]},"ISSN":["1477-7819"],"issn-type":[{"value":"1477-7819","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,11,9]]},"assertion":[{"value":"19 August 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"27 October 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 November 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Not applicable.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent participate"}},{"value":"Written informed consent was obtained from the patients for the publication of this case report and any accompanying images.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"292"}}