Pancreatic Cancer Secondary to Biliary Tract Cancer Associated with Pancreaticobiliary Maljunction: Intraductal Dissemination Through the Common Channel

Overview

Journal Clin J Gastroenterol

Specialty Gastroenterology

Date 2025 Mar 7

PMID 40053258

Authors

Keishi Sugimachi

Takahiro Tomino

Tomonari Shimagaki

Emi Onishi

Yutaka Koga

Kenichi Taguchi

Masayuki Furukawa

Terumasa Hisano

Rie Sugimoto

Masaru Morita

Affiliations

Soon will be listed here.

Abstract

Patients with pancreaticobiliary maljunction (PBM) have a high risk of biliary tract cancer (BTC). The risk of pancreatic cancer is also reported to be higher in patients with PBM compared to the general population; the underlying cause remains unclear. We report a 73-year-old man with widespread pancreatic cancer involving the entire pancreas. The patient previously underwent curative resection for gallbladder cancer and hilar cholangiocarcinoma concomitant with PBM. A total pancreatectomy was performed for the new pancreatic lesion. Histopathological examination revealed a papillary tumor predominantly composed of intraepithelial lesions that extended throughout the pancreatic duct, with skip lesions and irregular invasive foci at multiple sites within the duct. The morphological features and mucin profile were similar to those of the previous biliary lesions. Genetic analysis of the current lesion showed wild-type KRAS, GNAS, and PIK3CA genes consistent with the previous lesions, indicating that the pancreatic and biliary lesions were molecularly identical clones. Based on the clinicopathological findings and molecular analysis, we concluded that the BTC had spread intraluminally to the pancreatic duct through the common channel of the PBM, resulting in intraductal dissemination. Patients with PBM should be followed for pancreatic lesions, because of the risk of intraductal dissemination of BTC.

References

Kamisawa T, Honda G . Pancreaticobiliary Maljunction: Markedly High Risk for Biliary Cancer. Digestion. 2018; 99(2):123-125. DOI: 10.1159/000490816. View

Morine Y, Shimada M, Takamatsu H, Araida T, Endo I, Kubota M . Clinical features of pancreaticobiliary maljunction: update analysis of 2nd Japan-nationwide survey. J Hepatobiliary Pancreat Sci. 2013; 20(5):472-80. DOI: 10.1007/s00534-013-0606-2. View

Kamisawa T, Kuruma S, Chiba K, Tabata T, Koizumi S, Kikuyama M . Biliary carcinogenesis in pancreaticobiliary maljunction. J Gastroenterol. 2016; 52(2):158-163. DOI: 10.1007/s00535-016-1268-z. View

Ohuchida J, Chijiiwa K, Hiyoshi M, Kobayashi K, Konomi H, Tanaka M . Long-term results of treatment for pancreaticobiliary maljunction without bile duct dilatation. Arch Surg. 2006; 141(11):1066-70. DOI: 10.1001/archsurg.141.11.1066. View

Funabiki T, Matsubara T, Miyakawa S, Ishihara S . Pancreaticobiliary maljunction and carcinogenesis to biliary and pancreatic malignancy. Langenbecks Arch Surg. 2008; 394(1):159-69. DOI: 10.1007/s00423-008-0336-0. View

Kamisawa T, Takuma K, Anjiki H, Egawa N, Kurata M, Honda G . Pancreaticobiliary maljunction. Clin Gastroenterol Hepatol. 2009; 7(11 Suppl):S84-8. DOI: 10.1016/j.cgh.2009.08.024. View

Ueda N, Nagakawa T, Ohta T, Kayahara M, Ueno K, Konishi I . Synchronous cancer of the biliary tract and pancreas associated with anomalous arrangement of the pancreaticobiliary ductal system. J Clin Gastroenterol. 1992; 15(2):136-41. DOI: 10.1097/00004836-199209000-00011. View

Sato K, Maekawa T, Yabuki K, Tamasaki Y, Maekawa H, Kudo K . A case of triple synchronous cancers occurring in the gallbladder, common bile duct, and pancreas. J Gastroenterol. 2003; 38(1):97-100. DOI: 10.1007/s005350300014. View

Minami Y, Hasuike Y, Takeda Y, Tsujinaka T . Metachronous double cancer of the gallbladder and pancreas associated with pancreaticobiliary maljunction. J Hepatobiliary Pancreat Surg. 2008; 15(3):330-3. DOI: 10.1007/s00534-007-1208-7. View

10.

Lahmar A, Ben Abid S, Arfa M, Bayar R, Khalfallah M, Mzabi-Regaya S . Metachronous cancer of gallbladder and pancreas with pancreatobiliary maljunction. World J Gastrointest Surg. 2010; 2(4):143-6. PMC: 2999228. DOI: 10.4240/wjgs.v2.i4.143. View

11.

Rungsakulkij N, Boonsakan P . Synchronous gallbladder and pancreatic cancer associated with pancreaticobiliary maljunction. World J Gastroenterol. 2014; 20(39):14500-4. PMC: 4202380. DOI: 10.3748/wjg.v20.i39.14500. View

12.

Koizumi K, Sasajima J, Kawamoto T, Sugiyama Y, Muto M, Muto M . Multiple Cancers of the Biliary Tract and Pancreatic Duct after Cholecystectomy for Gallbladder Cancer in a Patient with Pancreaticobiliary Maljunction. Intern Med. 2016; 55(2):141-6. DOI: 10.2169/internalmedicine.55.4706. View

13.

Mori H, Iida H, Maehira H, Kitamura N, Shimizu T, Tani M . Synchronous primary gallbladder and pancreatic cancer associated with congenital biliary dilatation and pancreaticobiliary maljunction. Surg Case Rep. 2017; 3(1):113. PMC: 5668217. DOI: 10.1186/s40792-017-0388-x. View

14.

Sivade A, Sempoux C, Voutsadakis I, Brunel C, Halkic N, Godat S . Synchronous tumors of the pancreas and the gallbladder: a case report with targeted NGS evaluation. Ann Transl Med. 2020; 7(22):696. PMC: 6944562. DOI: 10.21037/atm.2019.10.107. View

15.

Kuhara Y, Hashimoto Y, Hirahara S, Kubota H, Shirakawa K, Toyota K . Triple metachronous primary pancreatic and gallbladder cancer associated with pancreaticobiliary maljunction: a case report. Surg Case Rep. 2021; 7(1):81. PMC: 8024430. DOI: 10.1186/s40792-021-01160-4. View

16.

Kato H, Asano Y, Ito M, Arakawa S, Shimura M, Koike D . A case of metachronous intraductal tubulopapillary carcinoma of the pancreas after surgery for gallbladder cancer-accompanied pancreaticobiliary maljunction. Clin J Gastroenterol. 2022; 15(5):1018-1025. DOI: 10.1007/s12328-022-01677-7. View

17.

Fujita N, NAKASE A . Experimental study of transductal dissemination of cancer of the pancreas. Gastroenterol Jpn. 1977; 12(3):224-9. DOI: 10.1007/BF02781767. View

18.

Yamaguchi R, Okabe Y, Jimi A, Shiota K, Kodama T, Naito Y . Pancreatic acinar cell carcinoma extending into the common bile and main pancreatic ducts. Pathol Int. 2006; 56(10):633-7. DOI: 10.1111/j.1440-1827.2006.02020.x. View

19.

Takahashi N, Taniguchi T, Adachi M . A case of needle tract seeding of an intraductal papillary neoplasm of the bile duct (IPNB) after percutaneous biopsy. Eur J Dermatol. 2014; 24(1):128-30. DOI: 10.1684/ejd.2013.2249. View

20.

Matsubara A, Nara S, Sekine S, Ojima H, Kosuge T, Shimada K . Intraductal dissemination of papillary adenocarcinoma of the ampulla of Vater in the pancreatic duct. Pathol Int. 2014; 64(1):39-44. DOI: 10.1111/pin.12131. View