Choledochal Cyst with Pancreas Divisum – Surgical Insight into A Rare Associatio
Abstract
Congenital pancreaticobiliary anomalies like pancreatic divisum (PD), choledochal cysts (CDC), anomalous pancreaticobiliary ductal union (APBDU) have been reported in 5.7% of patients undergoing magnetic resonance cholangiopancreatography. CDC’s are characterized by abnormal dilatations of the intrahepatic and/or extrahepatic portion of the biliary tree and can be complicated by cystolithiasis, cholangitis, pancreatitis and malignant transformation necessitating surgical managemnet. While CDCs are commonly associated with APBDU, combination of CDC with PD is rare and a potential surgical challenge. We report a case of recurrent cholangitis in a patient with CDC and coincidental classic PD and illustrate how preoperative identification, fastidious dissection technique to safeguard both the pancreatic ducts and simple intraoperative preemptive strategies may decrease consequences of distal stump blowout.
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[9] Hinojosa-Gonzalez DE, Roblesgil-Medrano A, Leon SUVD, Espadas-Conde MA, Flores-Villalba E. Biliary reconstruction after choledochal cyst resection: a systematic review and meta- analysis on hepaticojejunostomy vs hepaticoduodenostomy. Pediatr Surg Int. 2021 Oct;37(10):1313-22. https://doi. org/10.1007/s00383-021-04940-z [10] Lilly JR. The surgical treatment of choledochal cyst. Surg Gynecol Obstet. 1979 Jul;149(1):36-42.
[11] Liu Y, Sun J, Guo S, Liu Z, Zhu M, Zhang Z li. The distal classification and management of choledochal cyst in adults. Medicine (Baltimore). 2017 Mar;96(12):e6350. https://doi. org/10.1097/MD.0000000000006350
[12] Madadi-Sanjani O, Wirth TC, Kuebler JF, Petersen C, Ure BM. Choledochal Cyst and Malignancy: A Plea for Lifelong Follow- Up. Eur J Pediatr Surg. 2019 Apr;29(2):143-9. https://doi. org/10.1055/s-0037-1615275
[13] Xia HT, Yang T, Liang B, Zeng JP, Dong JH. Treatment and outcomes of adults with remnant intrapancreatic choledochal cysts. Surgery. 2016 Feb;159(2):418-25. https://doi. org/10.1016/j.surg.2015.04.042
[14] Dalvi AN, Pramesh CS, Prasanna GS, Rege SA, Khare R, Ravikiran CS. Incomplete pancreas divisum with anomalous choledochopancreatic duct junction with choledochal cyst. Arch Surg. 1999 Oct;134(10):1150-2. https://doi.org/10.1001/ archsurg.134.10.1150
[15] Anbarasu A, Deshpande A. Successful Management of a Post-Choledochal Cyst Excision Pancreatic Fistula in an Adult Patient: A Case Report and Literature Review on Risk Factors. Surg J (N Y). 2022 Jan;8(1):e41-5. https://doi. org/10.1055/s-0041-1742175
[16] Kolb A, Kleeff J, Frohlich B, Werner J, Friess H, Büchler MW. Resection of the intrapancreatic bile duct preserving the pancreas. J Hepatobiliary Pancreat Surg. 2009;16(1):31-4. https://doi.org/10.1007/s00534-008-0013-2
[17] Hackert T, Hartwig W, Werner J. Symptoms and Surgical Management of a Distal Choledochal Cyst in a Patient with Pancreas Divisum: Case Report and Review of the Literature. Case Rep Gastroenterol. 2007 Oct 2;1(1):90-5. https://doi.org/10.1159/000108636
[18] Ziegler KM, Pitt HA, Zyromski NJ, Chauhan A, Sherman S, Moffatt D, et al. Choledochoceles. Ann Surg. 2010 Oct;252(4):683-90. https://doi.org/10.1097/SLA.0b013e3181f6931f
[19] Law R, Topazian M. Diagnosis and treatment of choledochoceles. Clin Gastroenterol Hepatol. 2014 Feb;12(2):196-203. https:// doi.org/10.1016/j.cgh.2013.04.037
[20] McDonald ML, Farnell MB, Nagorney DM, Ilstrup DM, Kutch JM. Benign biliary strictures: repair and outcome with a contemporary approach. Surgery. 1995 Oct;118(4):582-90; discussion 590-1. doi: 10.1016/s0039-6060(05)80022-4. PMID: 7570309. https://doi.org/10.1016/S0039-6060(05)80022-4
[2] Ferri V, Vicente E, Quijano Y, Ielpo B, Duran H, Diaz E, et al. Diagnosis and treatment of pancreas divisum: A literature review. Hepatobiliary Pancreat Dis Int. 2019 Aug;18(4):332-6. https://doi.org/10.1016/j.hbpd.2019.05.004
[3] De Filippo M, Calabrese M, Quinto S, Rastelli A, Bertellini A, Martora R, et al. Congenital anomalies and variations of the bile and pancreatic ducts: magnetic resonance cholangiopancreatography findings, epidemiology and clinical significance. Radiol Med. 2008 Sep;113(6):841-59. https:// doi.org/10.1007/s11547-008-0298-x
[4] Soares KC, Kim Y, Spolverato G, Maithel S, Bauer TW, Marques H, et al. Presentation and Clinical Outcomes of Choledochal Cysts in Children and Adults. JAMA Surg. 2015 Jun 1;150(6):577. https://doi.org/10.1001/jamasurg.2015.0226
[5] Kamisawa T, Kaneko K, Itoi T, Ando H. Pancreaticobiliary maljunction and congenital biliary dilatation. Lancet Gastroenterol Hepatol. 2017 Aug;2(8):610-8. https://doi. org/10.1016/S2468-1253(17)30002-X
[6] Ransom-Rodríguez A, Blachman-Braun R, Sánchez-García Ramos E, Varela-Prieto J, Rosas-Lezama E, Mercado MÁ. A rare case of choledochal cyst with pancreas divisum: case presentation and literature review. Ann Hepatobiliary Pan- creat Surg. 2017 Feb;21(1):52-6. https://doi.org/10.14701/ ahbps.2017.21.1.52
[7] Pakkala A, Nagari B, Kumar Nekarakanti P, Kumar Bansal A. A case series of choledochal cyst with pancreatic divisum: A rare association. Turk J Surg. 2022 Sep 1;38(3):294-7. https://doi. org/10.47717/turkjsurg.2022.5609
[8] Ronnekleiv-Kelly SM, Soares KC, Ejaz A, Pawlik TM. Management of choledochal cysts. Curr Opin Gastroenterol. 2016;32(3):225- 31. https://doi.org/10.1097/MOG.0000000000000256
[9] Hinojosa-Gonzalez DE, Roblesgil-Medrano A, Leon SUVD, Espadas-Conde MA, Flores-Villalba E. Biliary reconstruction after choledochal cyst resection: a systematic review and meta- analysis on hepaticojejunostomy vs hepaticoduodenostomy. Pediatr Surg Int. 2021 Oct;37(10):1313-22. https://doi. org/10.1007/s00383-021-04940-z [10] Lilly JR. The surgical treatment of choledochal cyst. Surg Gynecol Obstet. 1979 Jul;149(1):36-42.
[11] Liu Y, Sun J, Guo S, Liu Z, Zhu M, Zhang Z li. The distal classification and management of choledochal cyst in adults. Medicine (Baltimore). 2017 Mar;96(12):e6350. https://doi. org/10.1097/MD.0000000000006350
[12] Madadi-Sanjani O, Wirth TC, Kuebler JF, Petersen C, Ure BM. Choledochal Cyst and Malignancy: A Plea for Lifelong Follow- Up. Eur J Pediatr Surg. 2019 Apr;29(2):143-9. https://doi. org/10.1055/s-0037-1615275
[13] Xia HT, Yang T, Liang B, Zeng JP, Dong JH. Treatment and outcomes of adults with remnant intrapancreatic choledochal cysts. Surgery. 2016 Feb;159(2):418-25. https://doi. org/10.1016/j.surg.2015.04.042
[14] Dalvi AN, Pramesh CS, Prasanna GS, Rege SA, Khare R, Ravikiran CS. Incomplete pancreas divisum with anomalous choledochopancreatic duct junction with choledochal cyst. Arch Surg. 1999 Oct;134(10):1150-2. https://doi.org/10.1001/ archsurg.134.10.1150
[15] Anbarasu A, Deshpande A. Successful Management of a Post-Choledochal Cyst Excision Pancreatic Fistula in an Adult Patient: A Case Report and Literature Review on Risk Factors. Surg J (N Y). 2022 Jan;8(1):e41-5. https://doi. org/10.1055/s-0041-1742175
[16] Kolb A, Kleeff J, Frohlich B, Werner J, Friess H, Büchler MW. Resection of the intrapancreatic bile duct preserving the pancreas. J Hepatobiliary Pancreat Surg. 2009;16(1):31-4. https://doi.org/10.1007/s00534-008-0013-2
[17] Hackert T, Hartwig W, Werner J. Symptoms and Surgical Management of a Distal Choledochal Cyst in a Patient with Pancreas Divisum: Case Report and Review of the Literature. Case Rep Gastroenterol. 2007 Oct 2;1(1):90-5. https://doi.org/10.1159/000108636
[18] Ziegler KM, Pitt HA, Zyromski NJ, Chauhan A, Sherman S, Moffatt D, et al. Choledochoceles. Ann Surg. 2010 Oct;252(4):683-90. https://doi.org/10.1097/SLA.0b013e3181f6931f
[19] Law R, Topazian M. Diagnosis and treatment of choledochoceles. Clin Gastroenterol Hepatol. 2014 Feb;12(2):196-203. https:// doi.org/10.1016/j.cgh.2013.04.037
[20] McDonald ML, Farnell MB, Nagorney DM, Ilstrup DM, Kutch JM. Benign biliary strictures: repair and outcome with a contemporary approach. Surgery. 1995 Oct;118(4):582-90; discussion 590-1. doi: 10.1016/s0039-6060(05)80022-4. PMID: 7570309. https://doi.org/10.1016/S0039-6060(05)80022-4
| Files | ||
| Issue | Vol 8 No 5 (2023): September-October | |
| Section | Case Report(s) | |
| DOI | https://doi.org/10.18502/crcp.v8i5.15264 | |
| Keywords | ||
| Choledochal cyst Pancreas divisum Fistula | ||
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How to Cite
1.
Kaw P, Behari A, Sharma S. Choledochal Cyst with Pancreas Divisum – Surgical Insight into A Rare Associatio. CRCP. 2024;8(5):223-227.
