Abstract
Choledochal cyst is a well-recognised entity, presenting primarily in infants and young children. Where symptoms are delayed until adulthood, associated hepatobiliary pathology may complicate the presentation. These problems may be aggravated by previous treatment with bypass surgery rather than resection. We report seven cases from our recent experience presenting with complications in adulthood. These included cholangitis, hepatic abscess, pancreatitis and malignancy within the cyst. Two patients presented during pregnancy. These complications and their implications for management are discussed.
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- ALONSO-LEJ F., REVER W. B., Jr, PESSAGNO D. J. Congenital choledochal cyst, with a report of 2, and an analysis of 94, cases. Int Abstr Surg. 1959 Jan;108(1):1–30. [PubMed] [Google Scholar]
- Adson M. A., Nagorney D. M. Hepatic resection for intrahepatic ductal stones. Arch Surg. 1982 May;117(5):611–616. doi: 10.1001/archsurg.1982.01380290067012. [DOI] [PubMed] [Google Scholar]
- Altman M. S., Halls J. M., Douglas A. P., Renner I. G. Choledochal cyst presenting as acute pancreatitis: evaluation with endoscopic retrograde cholangiopancreatography. Am J Gastroenterol. 1978 Nov;70(5):514–519. [PubMed] [Google Scholar]
- Angel J. L., Knuppel R. A., Trabin J. Choledochal cyst complicating a twin gestation. South Med J. 1985 Apr;78(4):463–466. doi: 10.1097/00007611-198504000-00025. [DOI] [PubMed] [Google Scholar]
- Austin R. M., Sussman S., McArdle C. R., Kim D., Elboim C. Computed tomographic and ultrasound appearances of a solitary intrahepatic choledochal cyst. Clin Radiol. 1986 Mar;37(2):149–150. doi: 10.1016/s0009-9260(86)80386-5. [DOI] [PubMed] [Google Scholar]
- Babbitt D. P., Starshak R. J., Clemett A. R. Choledochal cyst: a concept of etiology. Am J Roentgenol Radium Ther Nucl Med. 1973 Sep;119(1):57–62. doi: 10.2214/ajr.119.1.57. [DOI] [PubMed] [Google Scholar]
- Babbitt D. P. [Congenital choledochal cysts: new etiological concept based on anomalous relationships of the common bile duct and pancreatic bulb]. Ann Radiol (Paris) 1969;12(3):231–240. [PubMed] [Google Scholar]
- Barros J. L., Polo J. R., Sanabia J., Garcia-Sabrido J. L., Gomez-Lorenzo F. J. Congenital cystic dilatation of the intrahepatic bile ducts (Caroli's disease): report of a case and review of the literature. Surgery. 1979 May;85(5):589–592. [PubMed] [Google Scholar]
- Binstock M., Sondak V. K., Herd J., Reimnitz C., Lindsay K., Brinkman C., Roslyn J. J. Adenocarcinoma in a choledochal cyst during pregnancy: a case report and guidelines for management. Surgery. 1988 May;103(5):588–592. [PubMed] [Google Scholar]
- CAROLI J., SOUPAULT R., KOSSAKOWSKI J., PLOCKER L., PARADOWSKA La dilatation polykystique congénitale des voies biliaires intra-hépatiques; essai de classification. Sem Hop. 1958 Feb 18;34(8/2):488–95/SP. [PubMed] [Google Scholar]
- Cuschieri A., Davies R. S. Acute pancreatitis complicating a choledochal cyst. Br Med J. 1969 Sep 20;3(5672):698–698. doi: 10.1136/bmj.3.5672.698. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Dayton M. T., Longmire W. P., Jr, Tompkins R. K. Caroli's Disease: a premalignant condition? Am J Surg. 1983 Jan;145(1):41–48. doi: 10.1016/0002-9610(83)90164-2. [DOI] [PubMed] [Google Scholar]
- Diamond T., Panesar K. J. Biliary peritonitis due to choledochal cyst presenting in late pregnancy. Ulster Med J. 1986 Oct;55(2):190–192. [PMC free article] [PubMed] [Google Scholar]
- Flanigan D. P. Biliary carcinoma associated with biliary cysts. Cancer. 1977 Aug;40(2):880–883. doi: 10.1002/1097-0142(197708)40:2<880::aid-cncr2820400242>3.0.co;2-x. [DOI] [PubMed] [Google Scholar]
- Flanigan P. D. Biliary cysts. Ann Surg. 1975 Nov;182(5):635–643. doi: 10.1097/00000658-197511000-00017. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Fujiwara Y., Ohizumi T., Kakizaki G., Ishidate T. A case of congenital choledochal cyst associated with carcinoma. J Pediatr Surg. 1976 Aug;11(4):587–588. doi: 10.1016/s0022-3468(76)80021-8. [DOI] [PubMed] [Google Scholar]
- Glenn F., McSherry C. K. Congenital segmental cystic dilatation of the biliary ductal system. Ann Surg. 1973 Jun;177(6):705–713. doi: 10.1097/00000658-197306000-00009. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Jansen W. Intraduodenal cyst containing bile and stones: "choledochocele" in an accessory bile duct. Report of a case with recurrent pancreatitis. Gastroenterology. 1970 Mar;58(3):397–401. [PubMed] [Google Scholar]
- Jona J. Z., Babbitt D. P., Starshak R. J., LaPorta A. J., Glicklich M., Cohen R. D. Anatomic observations and etiologic and surgical considerations in choledochal cyst. J Pediatr Surg. 1979 Jun;14(3):315–320. doi: 10.1016/s0022-3468(79)80490-x. [DOI] [PubMed] [Google Scholar]
- Kagawa Y., Kashihara S., Kuramoto S., Maetani S. Carcinoma arising in a congenitally dilated biliary tract. Report of a case and review of the literature. Gastroenterology. 1978 Jun;74(6):1286–1294. [PubMed] [Google Scholar]
- Karjoo M., Bishop H. C., Borns P., Holtzapple P. G. Choledochal cyst presenting as recurrent pancreatitis. Pediatrics. 1973 Feb;51(2):289–291. [PubMed] [Google Scholar]
- Kitahama A., Harkness S. O., Moynihan P. O., Webb W. R. A large choledochal cyst with impending rupture post partum. Br J Surg. 1984 Feb;71(2):156–156. doi: 10.1002/bjs.1800710229. [DOI] [PubMed] [Google Scholar]
- Kotner L. M., Jr, McFadden J. C. Pancreatitis and choledochal cyst with gallstones: an unusual case. J Ark Med Soc. 1975 Mar;71(10):299–301. [PubMed] [Google Scholar]
- Longmire W. P., Jr, Mandiola S. A., Gordon H. E. Congenital cystic disease of the liver and biliary system. Ann Surg. 1971 Oct;174(4):711–726. doi: 10.1097/00000658-197110000-00014. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Matsumoto Y., Uchida K., Nakase A., Honjo I. Congenital cystic dilatation of the common bile duct as a cause of primary bile duct stone. Am J Surg. 1977 Sep;134(3):346–352. doi: 10.1016/0002-9610(77)90403-2. [DOI] [PubMed] [Google Scholar]
- Mercadier M., Chigot J. P., Clot J. P., Langlois P., Lansiaux P. Caroli's disease. World J Surg. 1984 Feb;8(1):22–29. doi: 10.1007/BF01658359. [DOI] [PubMed] [Google Scholar]
- Nagase M., Hikasa Y., Soloway R. D., Tanimura H., Setoyama M., Kato H. Gallstones in Western Japan. Factors affecting the prevalence of intrahepatic gallstones. Gastroenterology. 1980 Apr;78(4):684–690. [PubMed] [Google Scholar]
- Nagata E., Sakai K., Kinoshita H., Hirohashi K. Choledochal cyst: complications of anomalous connection between the choledochus and pancreatic duct and carcinoma of the biliary tract. World J Surg. 1986 Feb;10(1):102–110. doi: 10.1007/BF01656096. [DOI] [PubMed] [Google Scholar]
- Nagorney D. M., McIlrath D. C., Adson M. A. Choledochal cysts in adults: clinical management. Surgery. 1984 Oct;96(4):656–663. [PubMed] [Google Scholar]
- Okada A., Oguchi Y., Kamata S., Ikeda Y., Kawashima Y., Saito R. Common channel syndrome--diagnosis with endoscopic retrograde cholangiopancreatography and surgical management. Surgery. 1983 May;93(5):634–642. [PubMed] [Google Scholar]
- Ono J., Sakoda K., Akita H. Surgical aspect ot cystic dilatation of the bile duct. An anomalous junction of the pancreaticobiliary tract in adults. Ann Surg. 1982 Feb;195(2):203–208. doi: 10.1097/00000658-198202000-00014. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Powell C. S., Sawyers J. L., Reynolds V. H. Management of adult choledochal cysts. Ann Surg. 1981 May;193(5):666–676. doi: 10.1097/00000658-198105000-00018. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Pridgen J. E., Jr, Aust J. B., McInnis W. D. Primary intrahepatic gallstones. Arch Surg. 1977 Sep;112(9):1037–1043. doi: 10.1001/archsurg.1977.01370090019004. [DOI] [PubMed] [Google Scholar]
- Rattner D. W., Schapiro R. H., Warshaw A. L. Abnormalities of the pancreatic and biliary ducts in adult patients with choledochal cysts. Arch Surg. 1983 Sep;118(9):1068–1073. doi: 10.1001/archsurg.1983.01390090052012. [DOI] [PubMed] [Google Scholar]
- Stephens F. O., Pauline G. J. Choledochocele: an unusual type of choledochal cyst which presented as acute pancreatitis. Aust N Z J Surg. 1966 Nov;36(2):124–127. doi: 10.1111/j.1445-2197.1966.tb05505.x. [DOI] [PubMed] [Google Scholar]
- Stringel G., Filler R. M. Fictitious pancreatitis in choledochal cyst. J Pediatr Surg. 1982 Aug;17(4):359–361. doi: 10.1016/s0022-3468(82)80488-0. [DOI] [PubMed] [Google Scholar]
- Tan K. C., Howard E. R. Choledochal cyst: a 14-year surgical experience with 36 patients. Br J Surg. 1988 Sep;75(9):892–895. doi: 10.1002/bjs.1800750921. [DOI] [PubMed] [Google Scholar]
- Taylor T. V., Brigg J. K., Russell J. G., Torrance B. Choledochal cyst of pregnancy. J R Coll Surg Edinb. 1977 Nov;22(6):424–427. [PubMed] [Google Scholar]
- Todani T., Watanabe Y., Fujii T., Uemura S. Anomalous arrangement of the pancreatobiliary ductal system in patients with a choledochal cyst. Am J Surg. 1984 May;147(5):672–676. doi: 10.1016/0002-9610(84)90139-9. [DOI] [PubMed] [Google Scholar]
- Todani T., Watanabe Y., Mizuguchi T., Fujii T., Toki A. Hepaticoduodenostomy at the hepatic hilum after excision of choledochal cyst. Am J Surg. 1981 Nov;142(5):584–587. doi: 10.1016/0002-9610(81)90431-1. [DOI] [PubMed] [Google Scholar]
- Todani T., Watanabe Y., Narusue M., Tabuchi K., Okajima K. Congenital bile duct cysts: Classification, operative procedures, and review of thirty-seven cases including cancer arising from choledochal cyst. Am J Surg. 1977 Aug;134(2):263–269. doi: 10.1016/0002-9610(77)90359-2. [DOI] [PubMed] [Google Scholar]
- Trout H. H., 3rd, Longmire W. P., Jr Long-term follow-up study of patients with congenital cystic dilatation of the common bile duct. Am J Surg. 1971 Jan;121(1):68–86. doi: 10.1016/0002-9610(71)90080-8. [DOI] [PubMed] [Google Scholar]
- Tsuchida Y., Ishida M. Dilatation of the intrahepatic bile ducts in congenital cystic dilatation of the common bile duct. Surgery. 1971 May;69(5):776–781. [PubMed] [Google Scholar]
- Voyles C. R., Smadja C., Shands W. C., Blumgart L. H. Carcinoma in choledochal cysts. Age-related incidence. Arch Surg. 1983 Aug;118(8):986–988. doi: 10.1001/archsurg.1983.01390080088022. [DOI] [PubMed] [Google Scholar]
- Yamaguchi M. Congenital choledochal cyst. Analysis of 1,433 patients in the Japanese literature. Am J Surg. 1980 Nov;140(5):653–657. doi: 10.1016/0002-9610(80)90051-3. [DOI] [PubMed] [Google Scholar]
- Yamauchi S., Koga A., Matsumoto S., Tanaka M., Nakayama F. Anomalous junction of pancreaticobiliary duct without congenital choledochal cyst: a possible risk factor for gallbladder cancer. Am J Gastroenterol. 1987 Jan;82(1):20–24. [PubMed] [Google Scholar]
- Yue P. C. Choledochal cyst: a review of 18 cases. Br J Surg. 1974 Nov;61(11):896–900. doi: 10.1002/bjs.1800611109. [DOI] [PubMed] [Google Scholar]