Biliary Cancer Developed After the Reparative Surgery for Congenital Choledochal Cyst: a Case Report and Review of the Literature

Overview

Journal Int Cancer Conf J

Specialty Oncology

Date 2019 Jun 1

PMID 31149469

Authors

Kou Ikegame

Atsushi Takano

Hideki Watanabe

Atsushi Yamamoto

Yoshiaki Miyasaka

Kazushige Furuya

Haruka Nakada

Hidemitsu Sugai

Michiya Yasutome

Masayuki Inoue

Masao Hada

Hiroshi Nakagomi

Masao Omata

Toshio Oyama

Affiliations

Soon will be listed here.

Abstract

The incidence of bile duct cancer developed in the patients with congenital choledochal cysts (CC) with a history of reparative surgery is not well known. We experienced a case developed choleductal cancer 45 years after reparative surgery. She underwent reparative surgery with cyst excision and hepatic bile duct duodenal anastomosis at 1 year of age. She developed the symptoms of jaundice, anorexia, and dull pain in the right upper part of the abdomen at 47 years of age. The carcinoma arose from the dilated proximal bile duct anastomosed with the duodenum. Cholestasis and regurgitation of duodenal fluids seemed to have influenced the development of cancer in this patient. We additionally reviewed seven cohort studies concerning the incidence of biliary carcinoma after surgery for congenital choledocal cysts. The incidence of biliary cancer developed after surgery was 2.2 ± 2.5 (ranged 0-6.5)%. We also reviewed 33 Japanese case reports cited in Japan MEDLINE from 1986 to 2015. Regarding the CC types according to the Totani's classification, 12 were type I and 14 type IVa, 1 was type II and 6 were unknown type. The rate of coexistence of pancreaticobiliary maljunction (PBM) was 92%, (22/24; other 9 cases were not documented), and biliary cancer arose from the proximal stump of the reparative surgery in 68% (17/25) and from the distal stump in 32% (8/25) of cases. These findings suggested that the reflux of pancreatic juice due to PBM only partially explained the carcinogenesis of CCs. Intensive follow-up of such patients throughout their lives is necessary to avoid cancer death even after standard reparative surgery.

References

Eriguchi N, Aoyagi S, Okuda K, Hara M, Fukuda S, Tamae T . Carcinoma arising in the pancreas 17 years after primary excision of a choledochal cysts: report of a case. Surg Today. 2001; 31(6):534-7. DOI: 10.1007/s005950170117. View

Ishibashi T, Kasahara K, Yasuda Y, Nagai H, Makino S, Kanazawa K . Malignant change in the biliary tract after excision of choledochal cyst. Br J Surg. 1998; 84(12):1687-91. View

Watanabe Y, Toki A, Todani T . Bile duct cancer developed after cyst excision for choledochal cyst. J Hepatobiliary Pancreat Surg. 1999; 6(3):207-12. DOI: 10.1007/s005340050108. View

BABBITT D . [Congenital choledochal cysts: new etiological concept based on anomalous relationships of the common bile duct and pancreatic bulb]. Ann Radiol (Paris). 1969; 12(3):231-40. View

Ono S, Fumino S, Shimadera S, Iwai N . Long-term outcomes after hepaticojejunostomy for choledochal cyst: a 10- to 27-year follow-up. J Pediatr Surg. 2010; 45(2):376-8. DOI: 10.1016/j.jpedsurg.2009.10.078. View

Yoshikawa K, Yoshida K, Shirai Y, Sato N, Kashima Y, Coutinho D . A case of carcinoma arising in the intrapancreatic terminal choledochus 12 years after primary excision of a giant choledochal cyst. Am J Gastroenterol. 1986; 81(5):378-84. View

Yamamoto J, Shimamura Y, Ohtani I, Ohtani H, Yano M, Fukuda K . Bile duct carcinoma arising from the anastomotic site of hepaticojejunostomy after the excision of congenital biliary dilatation: a case report. Surgery. 1996; 119(4):476-9. DOI: 10.1016/s0039-6060(96)80151-6. View

Todani T, Watanabe Y, Toki A, Urushihara N . Carcinoma related to choledochal cysts with internal drainage operations. Surg Gynecol Obstet. 1987; 164(1):61-4. View

Fujisaki S, Akiyama T, Miyake H, Amano S, Tomita R, Fukuzawa M . A case of carcinoma associated with the remained intrapancreatic biliary tract 17 years after the primary excision of a choledochal cyst. Hepatogastroenterology. 1999; 46(27):1655-9. View

10.

Soares K, Kim Y, Spolverato G, Maithel S, Bauer T, Marques H . Presentation and Clinical Outcomes of Choledochal Cysts in Children and Adults: A Multi-institutional Analysis. JAMA Surg. 2015; 150(6):577-84. DOI: 10.1001/jamasurg.2015.0226. View

11.

Matsumoto Y, Fujii H, Itakura J, Matsuda M, Nobukawa B, Suda K . Recent advances in pancreaticobiliary maljunction. J Hepatobiliary Pancreat Surg. 2002; 9(1):45-54. DOI: 10.1007/s005340200004. View

12.

Kobayashi S, Asano T, Yamasaki M, Kenmochi T, Nakagohri T, Ochiai T . Risk of bile duct carcinogenesis after excision of extrahepatic bile ducts in pancreaticobiliary maljunction. Surgery. 1999; 126(5):939-44. DOI: 10.1016/s0039-6060(99)70036-x. View

13.

Ohashi T, Wakai T, Kubota M, Matsuda Y, Arai Y, Ohyama T . Risk of subsequent biliary malignancy in patients undergoing cyst excision for congenital choledochal cysts. J Gastroenterol Hepatol. 2012; 28(2):243-7. PMC: 3816325. DOI: 10.1111/j.1440-1746.2012.07260.x. View

14.

Takeshita N, Ota T, Yamamoto M . Forty-year experience with flow-diversion surgery for patients with congenital choledochal cysts with pancreaticobiliary maljunction at a single institution. Ann Surg. 2011; 254(6):1050-3. DOI: 10.1097/SLA.0b013e3182243550. View

15.

Flanigan D . Biliary carcinoma associated with biliary cysts. Cancer. 1977; 40(2):880-3. DOI: 10.1002/1097-0142(197708)40:2<880::aid-cncr2820400242>3.0.co;2-x. View

16.

KOMI N, Tamura T, Miyoshi Y, Kunitomo K, Udaka H, Takehara H . Nationwide survey of cases of choledochal cyst. Analysis of coexistent anomalies, complications and surgical treatment in 645 cases. Surg Gastroenterol. 1984; 3(2):69-73. View

17.

He X, Wang L, Liu W, Liu Q, Qu Q, Li B . The risk of carcinogenesis in congenital choledochal cyst patients: an analysis of 214 cases. Ann Hepatol. 2014; 13(6):819-26. View

18.

Kumamoto T, Tanaka K, Takeda K, Nojiri K, Mori R, Taniguchi K . Intrahepatic cholangiocarcinoma arising 28 years after excision of a type IV-A congenital choledochal cyst: report of a case. Surg Today. 2012; 44(2):354-8. PMC: 3898144. DOI: 10.1007/s00595-012-0387-2. View

19.

Katabi N, Pillarisetty V, DeMatteo R, Klimstra D . Choledochal cysts: a clinicopathologic study of 36 cases with emphasis on the morphologic and the immunohistochemical features of premalignant and malignant alterations. Hum Pathol. 2014; 45(10):2107-14. DOI: 10.1016/j.humpath.2014.06.016. View

20.

Shimamura K, Kurosaki I, Sato D, Takano K, Yokoyama N, Sato Y . Intrahepatic cholangiocarcinoma arising 34 years after excision of a type IV-A congenital choledochal cyst: report of a case. Surg Today. 2009; 39(3):247-51. DOI: 10.1007/s00595-008-3825-4. View