Indian J Surg (June 2013) 75(Suppl 1):S376–S378 DOI 10.1007/s12262-012-0512-x
CASE REPORT
Spontaneous Perforation of the Common Bile Duct in an Adult D. Paramhans & Sapna Shukla & Jitendra Grover
Received: 30 September 2008 / Accepted: 25 April 2012 / Published online: 9 August 2012 # Association of Surgeons of India 2012
Abstract Spontaneous perforation of the bile duct is a rare disease in children and even rarer in adults. Although diagnostic techniques have advanced over the past years, yet most cases are diagnosed at operation. A 44-year-old woman presented with epigastric pain and vomiting of one-day duration. Abdominal distension with tenderness and guarding was present all over the abdomen. Abdominal ultrasonography showed free fluid in the peritoneal cavity with sluggish peristalsis. Abdominal and chest radiographs were normal. At laparotomy, common bile duct CBD was perforated at the junction of CBD and cystic duct. Cholecystectomy with CBD exploration was done and CBD was closed over a T-tube, and external drainage was carried out. The patient recovered well, and on the 12th postoperative day, a T-tube cholangiogram was performed which was normal. The T-tube was removed on the 14th postoperative day. One month after operation, the patient was well. Examining the patient with a suspicious thinking helps in early diagnosis and management of the patient. Keywords Spontaneous . Common bile duct . Perforation
CBD perforation is uncommon, usually reported in infants due to congenital anomalies. Cases have been reported in adults following invasive procedures in and around the CBD. But spontaneous perforation of CBD occurring in an adult presenting as acute abdomen is an unusual phenomenon. We report one such case which presented as acute abdomen exploratory. Laparotomy was done and CBD was D. Paramhans : S. Shukla : J. Grover (*) Department of Surgery, MGM Medical College and MY Hospital, Flat No 303, Gyatari Apartments 111,112 Geeta Nagar, Indore, India e-mail: [email protected]
found perforated. CBD was explored and closed over the T-tube and the patient recovered uneventfully (Fig. 1).
Case A 44-year-old woman presented with epigastric pain and vomiting of one-day duration. On examination, the abdomen was distended. There was tenderness and guarding in the whole abdomen. On ultrasound examination, there was moderate free fluid in the peritoneal cavity with sluggish peristalsis. Plain radiograph abdomen and chest were normal. Investigations of complete hemogram, blood urea, serum creatinine, and serum electrolytes were within normal limits. The patient was resuscitated and explored. There was bile mixed with peritoneal fluid in the peritoneal cavity. Whole of small and large bowel was explored and was found normal. A perforation was found in the common bile duct (CBD) near the junction of cystic duct and CBD. Cholecystectomy was done and the CBD was explored. There was no calculus, stenosis, or any malformation of CBD. The CBD was closed over a T-tube. Peritoneal lavage was given and abdomen was closed with a subhepatic drain. Bile culture showed Escherichia coli and Widal was also within normal limits. Patient’s ELISA for HIV was normal. Investigations for Mycobacterium tuberculosis were negative. The patient had a smooth recovery in the post-operative period. AT-tube cholangiogram was performed on the 12th post-operative day, which was normal. The T-tube was removed on the 14th postoperative day. The patient was discharged after removal of the T-tube.
Discussion Spontaneous CBD perforations are more commonly reported in infants. The etiological factors that are thought
Indian J Surg (June 2013) 75(Suppl 1):S376–S378
S377
Perforation is also found in patients after biliary surgery as a result of the following: (i) (ii)
Sloughing of the cystic duct stump [19] Weakness of the duct wall after previous choledochotomy [12] (iii) Spontaneous perforation due to retained or newly formed stones [12] (iv) After ERCP in 0.1–1.8 % of cases [20]
Fig. 1 Closure of CBD over T‐tube
to be the cause of spontaneous CBD perforation in infants are choledochal cysts and anomalous union of the pancreaticobiliary ductal system which results in reflux of pancreatic juices and tissue destruction, distal bile duct stenosis, or atresia leading to congenital weakness of the CBD [1–5]. Spontaneous bile duct perforation in other age groups may be due to single or multiple factors: (i)
Increased intraductal pressure by gallstones [3, 6, 7], mucus plugs [8], tumor [9], parasites [10], acquired or congenital strictures of the biliary tree [11], spasm or disease of the sphincter of Oddi, or alteration of the ampulla of Vater [12, 13] (ii) Erosion or necrosis of the bile duct wall due to compression by stones [3] (iii) Infection and thrombosis of intramural vessels with ischemic necrosis in the case of cholangitis [14] or suppuration with abscess formation around the bile duct [15] (iv) Malformations such as diverticula and congenital cystic dilatation [11, 16, 17] (v) Congenital malfunction of the wall at the junction of the cystic and common hepatic ducts [18], although this has been questioned [6, 14]
Perforation of the common bile duct was related to the abrupt increase in intraluminal pressure. The sudden increase in the intraluminal pressure also results in decreased blood flow in the vessels which run along the lateral border of the bile ducts, resulting in ischemia on the anterior surface of the bile duct [21]. Common bile duct perforation may be as a result of infection with (i) Mycobacteriun tuberculosis [22] or (ii) Salmonella typhi [23]. The commonest reported site of perforation is at the junction of the cystic and hepatic ducts [4, 5, 24]. Other sites include cystic duct, common hepatic duct, and common bile duct. Recommended treatment for such cases is T-tube drainage of the common bile duct along with cholecystectomy. In cases with distal obstruction of the CBD, a biliary enteric bypass should be done. Primary suture repair of the CBD is considered if a preoperative cholangiogram is available and it shows no pathology distal to the perforation. If preoperative cholangiogram is not available, then the condition is best managed by closure over a T-tube [25]. In conclusion, spontaneous perforation of the extrahepatic bile duct is a rare but important condition in adults. If not diagnosed timely, it can be a cause of mortality; thus, having knowledge of this condition helps in early diagnosis and management of the patient.
References 1. Ando K, Miyono T, Kohno S et al (1999) Spontaneous perforation of choledochal cyst: a study of 13 cases. Eur J Pediatr Surg 8(l):23–25 2. Treem WR, Hyams JS, McGowan GS et al (1991) Spontaneous rupture of a choledochal cyst: clues to diagnosis and etiology. J Pediatr Gastroenterol Nutr 13(3):301–306 3. Gatiepy JS, Capano OA, Gardner LWM (1951) Non-traumatic rupture of the common bile duct. Am J Surg 81:357–362 4. Hasegawa T, Udatsu Y, Kamiyama M et al (2000) Does pancreatico-biliary maljunction play a role in spontaneous perforation of the bile duct in children? Pediatr Surg Int 16:550–553 5. Stringel G, Mercer S (1983) Idiopathic perforation of the biliary tract in infancy. J Pediatr Surg 18:546–550 6. Hart EE (1951) Spontaneous perforation of the common bile duct. Ann Surg 133:280–282 7. Gough AZ, Edwards AN, Kedclie NE (1976) Spontaneous perforation of the common hepatic duct. Br J Surg 63:446–448 8. Newell ED (1941) Spontaneous rupture of the common bile duct. Ann Surg 113:877–880
S378 9. Spira IA (1976) Spontaneous rupture of the common bile duct. Ann Surg 183:433–435 10. Wig JD, Kaushir SP, Saleem MA, Talwar BL (1983) Spontaneous perforation of common bile duct. R Coll Surg Edinb 28:240–243 11. Gertz TE (1959) Spontaneous perforation of the common bile duet. Arch Surg 78:7–11 12. Igini JP, Fox PF (1966) Spontaneous perforation of the common bile duct. Am Surg 111:745–748 13. Pliteri S, Liberti E, Castelli E, Guccarine G (1972) Un cas rare de perforation Chole’docienne par calcul. Lyon Chir 68:127–129 14. Soarez I, Detrner DE, Jarret F (1981) Surgical management of spontaneous hepatic duct perforations. Ann Surg 194:176–179 15. Plehne WE, De G (1978) Spontaneous perforation of the common bile duct. Aust N Z J Surg 48:557–558 16. Chen WJ, Chang CH, Hung WT (1973) Congenital choledochal cyst: with observations on rupture of the cyst and intrahepatic ductal dilatation. J Pediatr Surg 8:529–538 17. Jackson BT, Sanders P (1971) Perforated choledochal cyst. Br J Surg 58:38–42
Indian J Surg (June 2013) 75(Suppl 1):S376–S378 18. Lilley J (1974) Spontaneous perforation of the extra hepatic bile duets and bile peritonitis in infancy. Surgery 75:664–673 19. Steinboff NG, Tucker GL (1971) Non-traumatic perforation of the commonduct. Am J Surg 121:334–337 20. Lai CH, Lau WY (2008) Management of ERCP related perforation. Surgeon 6(1):45–48 21. Ando H, Ito T, Watanabe Y et al (1995) Spontaneous perforation of choledochal cyst. J Am Coll Surg 181(2):125–128 22. Jarmin R, Imran R (2004) Common bile duct perforation due to tuberculosis. Asian J Surg 27(4):342–415 23. Gosbell I, Jones PD, Matthews A, Yeo B (1995) Surgical presentation of Hepatobiliary disease due to Salmonella typhi. Aust N Z J Surg 65(12):898–899 24. Chardot C, Iskandarani F, De Dreuzy O et al (1996) Spontaneous perforation of the biliary tract in infancy: a series of 11 cases. Eur J Pediatr Surg 6:341–346 25. Spigland N, Greco R, Rosenfeld D (1983) Spontaneous biliary perforation. Does ext drainage constitute adequate therapy? J Pediatr Surg 18:546–550
CASE REPORT
Spontaneous Perforation of the Common Bile Duct in an Adult D. Paramhans & Sapna Shukla & Jitendra Grover
Received: 30 September 2008 / Accepted: 25 April 2012 / Published online: 9 August 2012 # Association of Surgeons of India 2012
Abstract Spontaneous perforation of the bile duct is a rare disease in children and even rarer in adults. Although diagnostic techniques have advanced over the past years, yet most cases are diagnosed at operation. A 44-year-old woman presented with epigastric pain and vomiting of one-day duration. Abdominal distension with tenderness and guarding was present all over the abdomen. Abdominal ultrasonography showed free fluid in the peritoneal cavity with sluggish peristalsis. Abdominal and chest radiographs were normal. At laparotomy, common bile duct CBD was perforated at the junction of CBD and cystic duct. Cholecystectomy with CBD exploration was done and CBD was closed over a T-tube, and external drainage was carried out. The patient recovered well, and on the 12th postoperative day, a T-tube cholangiogram was performed which was normal. The T-tube was removed on the 14th postoperative day. One month after operation, the patient was well. Examining the patient with a suspicious thinking helps in early diagnosis and management of the patient. Keywords Spontaneous . Common bile duct . Perforation
CBD perforation is uncommon, usually reported in infants due to congenital anomalies. Cases have been reported in adults following invasive procedures in and around the CBD. But spontaneous perforation of CBD occurring in an adult presenting as acute abdomen is an unusual phenomenon. We report one such case which presented as acute abdomen exploratory. Laparotomy was done and CBD was D. Paramhans : S. Shukla : J. Grover (*) Department of Surgery, MGM Medical College and MY Hospital, Flat No 303, Gyatari Apartments 111,112 Geeta Nagar, Indore, India e-mail: [email protected]
found perforated. CBD was explored and closed over the T-tube and the patient recovered uneventfully (Fig. 1).
Case A 44-year-old woman presented with epigastric pain and vomiting of one-day duration. On examination, the abdomen was distended. There was tenderness and guarding in the whole abdomen. On ultrasound examination, there was moderate free fluid in the peritoneal cavity with sluggish peristalsis. Plain radiograph abdomen and chest were normal. Investigations of complete hemogram, blood urea, serum creatinine, and serum electrolytes were within normal limits. The patient was resuscitated and explored. There was bile mixed with peritoneal fluid in the peritoneal cavity. Whole of small and large bowel was explored and was found normal. A perforation was found in the common bile duct (CBD) near the junction of cystic duct and CBD. Cholecystectomy was done and the CBD was explored. There was no calculus, stenosis, or any malformation of CBD. The CBD was closed over a T-tube. Peritoneal lavage was given and abdomen was closed with a subhepatic drain. Bile culture showed Escherichia coli and Widal was also within normal limits. Patient’s ELISA for HIV was normal. Investigations for Mycobacterium tuberculosis were negative. The patient had a smooth recovery in the post-operative period. AT-tube cholangiogram was performed on the 12th post-operative day, which was normal. The T-tube was removed on the 14th postoperative day. The patient was discharged after removal of the T-tube.
Discussion Spontaneous CBD perforations are more commonly reported in infants. The etiological factors that are thought
Indian J Surg (June 2013) 75(Suppl 1):S376–S378
S377
Perforation is also found in patients after biliary surgery as a result of the following: (i) (ii)
Sloughing of the cystic duct stump [19] Weakness of the duct wall after previous choledochotomy [12] (iii) Spontaneous perforation due to retained or newly formed stones [12] (iv) After ERCP in 0.1–1.8 % of cases [20]
Fig. 1 Closure of CBD over T‐tube
to be the cause of spontaneous CBD perforation in infants are choledochal cysts and anomalous union of the pancreaticobiliary ductal system which results in reflux of pancreatic juices and tissue destruction, distal bile duct stenosis, or atresia leading to congenital weakness of the CBD [1–5]. Spontaneous bile duct perforation in other age groups may be due to single or multiple factors: (i)
Increased intraductal pressure by gallstones [3, 6, 7], mucus plugs [8], tumor [9], parasites [10], acquired or congenital strictures of the biliary tree [11], spasm or disease of the sphincter of Oddi, or alteration of the ampulla of Vater [12, 13] (ii) Erosion or necrosis of the bile duct wall due to compression by stones [3] (iii) Infection and thrombosis of intramural vessels with ischemic necrosis in the case of cholangitis [14] or suppuration with abscess formation around the bile duct [15] (iv) Malformations such as diverticula and congenital cystic dilatation [11, 16, 17] (v) Congenital malfunction of the wall at the junction of the cystic and common hepatic ducts [18], although this has been questioned [6, 14]
Perforation of the common bile duct was related to the abrupt increase in intraluminal pressure. The sudden increase in the intraluminal pressure also results in decreased blood flow in the vessels which run along the lateral border of the bile ducts, resulting in ischemia on the anterior surface of the bile duct [21]. Common bile duct perforation may be as a result of infection with (i) Mycobacteriun tuberculosis [22] or (ii) Salmonella typhi [23]. The commonest reported site of perforation is at the junction of the cystic and hepatic ducts [4, 5, 24]. Other sites include cystic duct, common hepatic duct, and common bile duct. Recommended treatment for such cases is T-tube drainage of the common bile duct along with cholecystectomy. In cases with distal obstruction of the CBD, a biliary enteric bypass should be done. Primary suture repair of the CBD is considered if a preoperative cholangiogram is available and it shows no pathology distal to the perforation. If preoperative cholangiogram is not available, then the condition is best managed by closure over a T-tube [25]. In conclusion, spontaneous perforation of the extrahepatic bile duct is a rare but important condition in adults. If not diagnosed timely, it can be a cause of mortality; thus, having knowledge of this condition helps in early diagnosis and management of the patient.
References 1. Ando K, Miyono T, Kohno S et al (1999) Spontaneous perforation of choledochal cyst: a study of 13 cases. Eur J Pediatr Surg 8(l):23–25 2. Treem WR, Hyams JS, McGowan GS et al (1991) Spontaneous rupture of a choledochal cyst: clues to diagnosis and etiology. J Pediatr Gastroenterol Nutr 13(3):301–306 3. Gatiepy JS, Capano OA, Gardner LWM (1951) Non-traumatic rupture of the common bile duct. Am J Surg 81:357–362 4. Hasegawa T, Udatsu Y, Kamiyama M et al (2000) Does pancreatico-biliary maljunction play a role in spontaneous perforation of the bile duct in children? Pediatr Surg Int 16:550–553 5. Stringel G, Mercer S (1983) Idiopathic perforation of the biliary tract in infancy. J Pediatr Surg 18:546–550 6. Hart EE (1951) Spontaneous perforation of the common bile duct. Ann Surg 133:280–282 7. Gough AZ, Edwards AN, Kedclie NE (1976) Spontaneous perforation of the common hepatic duct. Br J Surg 63:446–448 8. Newell ED (1941) Spontaneous rupture of the common bile duct. Ann Surg 113:877–880
S378 9. Spira IA (1976) Spontaneous rupture of the common bile duct. Ann Surg 183:433–435 10. Wig JD, Kaushir SP, Saleem MA, Talwar BL (1983) Spontaneous perforation of common bile duct. R Coll Surg Edinb 28:240–243 11. Gertz TE (1959) Spontaneous perforation of the common bile duet. Arch Surg 78:7–11 12. Igini JP, Fox PF (1966) Spontaneous perforation of the common bile duct. Am Surg 111:745–748 13. Pliteri S, Liberti E, Castelli E, Guccarine G (1972) Un cas rare de perforation Chole’docienne par calcul. Lyon Chir 68:127–129 14. Soarez I, Detrner DE, Jarret F (1981) Surgical management of spontaneous hepatic duct perforations. Ann Surg 194:176–179 15. Plehne WE, De G (1978) Spontaneous perforation of the common bile duct. Aust N Z J Surg 48:557–558 16. Chen WJ, Chang CH, Hung WT (1973) Congenital choledochal cyst: with observations on rupture of the cyst and intrahepatic ductal dilatation. J Pediatr Surg 8:529–538 17. Jackson BT, Sanders P (1971) Perforated choledochal cyst. Br J Surg 58:38–42
Indian J Surg (June 2013) 75(Suppl 1):S376–S378 18. Lilley J (1974) Spontaneous perforation of the extra hepatic bile duets and bile peritonitis in infancy. Surgery 75:664–673 19. Steinboff NG, Tucker GL (1971) Non-traumatic perforation of the commonduct. Am J Surg 121:334–337 20. Lai CH, Lau WY (2008) Management of ERCP related perforation. Surgeon 6(1):45–48 21. Ando H, Ito T, Watanabe Y et al (1995) Spontaneous perforation of choledochal cyst. J Am Coll Surg 181(2):125–128 22. Jarmin R, Imran R (2004) Common bile duct perforation due to tuberculosis. Asian J Surg 27(4):342–415 23. Gosbell I, Jones PD, Matthews A, Yeo B (1995) Surgical presentation of Hepatobiliary disease due to Salmonella typhi. Aust N Z J Surg 65(12):898–899 24. Chardot C, Iskandarani F, De Dreuzy O et al (1996) Spontaneous perforation of the biliary tract in infancy: a series of 11 cases. Eur J Pediatr Surg 6:341–346 25. Spigland N, Greco R, Rosenfeld D (1983) Spontaneous biliary perforation. Does ext drainage constitute adequate therapy? J Pediatr Surg 18:546–550
