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Biliary complications after excisional procedure for choledochal cyst
Biliary Complications After Excisional Procedure for Choledochal Cyst By T. Todani, Y. Watanabe, N. Urushihara, T. Noda, and Y. Morotomi
Kagawa, Japan • During the last 25 years, from 1969 to 1994, the authors treated 97 choledochal cysts by surgical excision. Biliary reconstruction consisted of 67 hepaticoduodenostomies and 30 hepaticojejunostomies. The common hepatic duct was the site of anastomosis in 9 of the duodenostomies and 13 of the jejunostomies and of the bifurcation of the hepatic ducts in 58 duodenostomies and 17 jejunostomies. Reoperation was required in 10 cases because of recurrent cholangitis with intrahepatic gallstones. Biliary strictures were responsible for the cholangitis in 9 children with anastomoses at the level of the common hepatic duct and in 1 with an anastomosis at the level of the bifurcation. These results suggest that biliary complications develop because of anastomotic stricture or primary ductal stricture, and may be minimised by the creation of a wide anastomosis, which is best accomplished at the hepatic hilum.
Copyright © 1995 by W.B. Saunders Company INDEX WORDS: Choledochal cyst; anastomotic stricture; primary ductal stricture; cholangitis.
XCISIONAL procedure for choledochal cyst is
now recognised as the treatment of choice. E Lesser procedures such as cystoduodenostomy have
been associated with high morbidity rates and with the potential for malignant change in the biliary tree. 1-4 Excision also has the advantage of stopping reflux of pancreatic juice through pancreaticobiliary maljunction, which is so often associated with choledochal cysts.5 However, recurrent ascending cholangitis and intrahepatic stone formation has necessitated reoperation in 10 of our patients who had previously undergone cyst excision. This article reviews the late complications of excisional surgery and compares the methods of bilioenteric anastomoses. MATERIALS AND METHODS Choledochal cyst excision was performed in 97 patients between February 1969 and April 1994 (Table 1). Reconstruction was performed with either a hepaticoduodenostomy or a Roux-en-Y jejunostomy, and the anastomoses were either below or at the hilum of the hepatic ducts. 1,6Ages ranged from 27 days to 37 years
From the Department of Pediatric Surgery, Kagawa Medical School, Kagawa, Japan. Presented at the 41st Annual International Congress of the British Association of Paediatric Surgeons, Rotterdam, The Netherlands, June 29-July 1, 1994. Address reprint requests to Takuji Todani, MD, Department of Pediatric Surgery, Kagawa Medical School, 1750-1 Miki, Kitagun, Kagawa 761-07, Japan. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3003-0025503. 00/0 478
Table 1. Primary Reconstruction Methods, February 1969 to April 1994 Hepaticoduodenostomy
Hepaticojejujnostomy
At Hilum
At Hilum
Below Hilum
Below Hilum
Type [n]
I [45]
25
5
IV [52] Subtotal
33 58
4 (4) 9 (4)
Total
9
6 (3)
8 (1) 17 (1)
67 (4)
7 (2) 13 (5) 30 (6)
NOTE: I, cystic or diffuse choledochal dilatation; IV, intrahepatic involvement; parentheses indicate number of reoperated patients.
(Table 2), and there was no evidence of malignant change in any of the cysts. Hepaticoenterostomies distal to the hilum were performed in the first 22 patients, and these consisted of hepaticoduodenostomies in 9 cases and hepaticojejunostomies in 13 (Fig 1). One of these cases required reoperation for recurrent cholangitis 7 years after the primary procedure, 6 and 75 subsequent cases were therefore treated with more proximal anastomoses at the level of the hepatic hilum in an attempt to provide a wider anastomosis. In this latter group, hepaticoduodenostomy was the procedure of choice in 58 and Roux-en-Y jejunostomy in 17 cases (Fig 1). RESULTS
Nine of the 22 patients treated with hepaticoenterostomy distal to the hilum required reoperation several years after surgery because of recurrent chop angitis with intrahepatic cholelithiasis secondary to anastomotic stricture formation (Fig 1, Table 3). Five of these patients had undergone primary surgery in our department, and 4 were referred from elsewhere. Late stricturing of hepaticoenteric anastomosis is illustrated by the case of a 9-year-old girl (case 2) who underwent excision for type I cyst and an end-to-side Roux-en-Y hepaticojejunostomy distal to the hilum. The anastomosis measured 16 mm. Episodes of pyrexia and abdominal pain occurred 12 years after surgery. A percutaneous transhepatic cholangiogram Table 2. Age Distribution Age
n
0-12 months 1-5 years 6-10,years 11-20 years 21 years+
25
Total
97
21 17 21 13
Journal of Pediatric Surgery, Vo130, No 3 (March), 1995: pp 478-481
COMPLICATIONS OF CHOLEDOCHAL CYST EXCISION BELOWHILUM
479
AT HILUM
¢Jtt hepaticoduodenostemy
hepatieoj ejunostomy with blind pouch
hepaticoduodenostomy
hepatico jejunostomy end-to-end fashion
13 (5)
22 (9)
"~
~
k
97 (10) ~ Fig 1. Biliary reconstructions performed from February 1969 to April 1994. Numbers in parentheses indicate the number of patients who underwent reoperation.
showed an anastomotic stricture 4 mm in diameter and intrahepatic gallstone formation (Fig 2). Laparotomy showed atrophy of the right lobe of the liver and multiple gallstones. Treatment consisted of right hepatectomy and reanastomosis with an end-to-end hepaticojejunostomy at the hilum. Postoperative progress has been very satisfactory for 3 years. Seventy-four of 75 patients who have undergone hepaticoenterostomy at the hilum with a wide anastomosis have achieved favorable results. The exception was a 17-year-old girl (case 10) who underwent excision for type IV cyst and an end-to-end Rouxen-Y hepaticojejunostomy at the hilum. An intraoperative cholangiogram showed some stricturing of the left hepatic duct near the hilum (Fig 3A). Pyrexia, jaundice, and abdominal pain recurred 15 years later, and a percutaneous transhepatic cholangiogram re-
Fig 2. Case 2. A cholangiogram when patient was 30 years of age taken through PTCD (percutaneous transhepatic cholangio drainage) tube showing anastomotic stricture associated with multiple stones in the hilum and the posterior ducts of the right hepatic lobe,
vealed a severe stricture of the left hepatic duct, possibly a result of a previous incomplete plasty of the hilar duct (Fig 3B). Operation showed no anastomotic stricture but atrophy of the lateral left hepatic
Table 3. Case Summary of Reoperation Age (yr)
Redo-Operation
Age (yr)
Follow-Up(yr)
Anastomotic stricture 1. Hepaticojejunostomy* 2. Hepaticojejunostomy
Type
10 9
15 30
4 3
Hepaticojejunostomy*
16
Hepaticoduodenostomy Hepaticojejunostomy* Hepaticoduodenostomy
11 2 5
23 26 18 6 11
7 2 17 7 6
13 18 15
Hepaticojejunostomy Hepaticojejunostomy and right hepatectomy Hepaticojejunostomy* Hepaticojejunostomy Hepaticojejunostomy Hepaticoduodenostomy Lateral segmentectomy with hepaticojejunostomy Hepaticojejunostomy Hepaticojejunostomy* Hepaticojejunostomy
28 33 24
5 4 3
17
Lateral segmentectomy
32
3
4. IV 5. IV 6. IV
First Operation
7. IV Hepaticojejunostomy 8. IV Hepaticoduodenostomy 9. IV Hepaticoduodenostomy* Primary ductal stricture 10. IV Hepaticojejunostomy *Performed elsewhere.
480
TODANI ET AL
Fig 3. Case 10. (A) An operative cholangiogram at the patient age of 17 years indicating a slight stricture of the left hepatic duct near the hepatic hilum. (B) A cholangiogram when patient was 32 years of age taken through PTC tube showing severe stricture of the left hepatic duct near the hilum with intrahepatic gallstones,
segment. A lateral segmentectomy resulted in a resolution of the symptoms. DISCUSSION
Recurrent symptoms of cholangitis after choledochal cyst excision may be the result of either a stricture at the site of anastomosis or a primary ductal stricture. 6-8 In our series there has been no significant difference in the occurrence rate of cholangitis between hepaticoduodenostomy and Roux-en-Y jejunostomy. 7,8 We have favoured hepaticoduodenostomy because we believe that the operation is more physiological, it is simpler to perform, and there are less complications from adhesive obstruction, anastomotic leakage, and peptic ulceration. 6 Cholangitis secondary to anastomotic stricture has been more frequent in patients who have undergone operations with anastomoses distal to the bifurcation of the hepatic duct and in those with intrahepatic ductal dilatation (type IV cyst). In the group with type IV cysts, anastomotic stricture developed in all four patients with hepaticoduodenostomies performed distal to the hilum and in two of seven of those with jejunostomies anastomosed at similar levels (Table 1). The reasons for this complication may include the
small diameter of the common hepatic duct in many cases of choledochal cyst, a relatively poor blood supply, and epithelial damage and intramural fibrosis. Primary strictures of the hepatic ducts near hilum and intrahepatic ducts are occasionally seen in patients with choledochal cysts, and whenever possible these should be widened by ductoplasties at the time of the primary surgery. Intrahepatic duct strictures may respond to repeated dilatation, although occasionally a segmental hepatic resection may be necessary. A hilar anastomosis allows the construction of a wide anastomosis, which may be performed with a continuous suture, and the chance of anastomotic leakage are lessened. 9 Wide anastomoses are essential to prevent the complication of ascending cholangitis and can be achieved by extending the incisions along the lateral walls of the hepatic ducts 6,1° (Fig 1). This is essential for type IV cysts with intrahepatic involvement. Provided a wide anastomosis was achieved in our series, favorable results could be obtained in both the duodenostomy and the jejunostomy groups. We therefore suggest that surgical enlargement of the hilar ducts to provide wide stoma should be routine in all patients undergoing choledochal cyst excision.
REFERENCES 1. Todani T, Watanabe Y, Narusue M, et al: Congenital bile duct cyst; its classification, operative procedures, and review of 37 cases including cancer arising from choledochal cyst. Am J Surg 134:263-269, 1977 2. Lilly JR: The surgical treatment of choledochal cyst. Surg Gynecol Obstet 149:36-42, 1979
3. Deziel DJ, Rossi RL, Munson L, et al: Management of bile duct cysts in adults. Arch Surg 121:410-415, 1986 4. Yoshikawa K, Yoshida K, Shirai Y, et al: A case of carcinoma arising in the intrapancreatic terminal choledochus 12 years after primary excision of a giant choledochal cyst. Am J Gastroenterol 81:378-384, 1986
COMPLICATIONS OF CHOLEDOCHAL CYST EXCISION
5. Todani T, Watanabe Y, Fujii T, et al: Anomalous arrangement of the pancreatobiliary ductal system in patients with a choledochal cyst. Am J Surg 147:672-676, 1984 6. Todani T, Narusue M, Watanabe Y, et al: Management of congenital choledochal cyst with intrahepatic involvement. Ann Surg 187:272-280, 1978 7. Todani T, Watanabe Y, Toki A, et al: Reoperation for congenital choledochal cyst. Ann Surg 207:142-147, 1988
481
8. Lilly JR: Surgery of coexisting biliary malformations in choledochal cyst. J Pediatr Surg 14:643-647, 1979 9. Terblanche J, Worthley CS, Spence RAJ, et al: High or low hepatico-jejunostomy for bile duct stricture? 108:828-834, 1990 10. Todani T, Watanabe Y, Mizuguchi T, et al: Hepaticoduodenostomy at the hepatic hilum after excision of choledochal cyst. Am J Surg 142:584-581, 1981
Kagawa, Japan • During the last 25 years, from 1969 to 1994, the authors treated 97 choledochal cysts by surgical excision. Biliary reconstruction consisted of 67 hepaticoduodenostomies and 30 hepaticojejunostomies. The common hepatic duct was the site of anastomosis in 9 of the duodenostomies and 13 of the jejunostomies and of the bifurcation of the hepatic ducts in 58 duodenostomies and 17 jejunostomies. Reoperation was required in 10 cases because of recurrent cholangitis with intrahepatic gallstones. Biliary strictures were responsible for the cholangitis in 9 children with anastomoses at the level of the common hepatic duct and in 1 with an anastomosis at the level of the bifurcation. These results suggest that biliary complications develop because of anastomotic stricture or primary ductal stricture, and may be minimised by the creation of a wide anastomosis, which is best accomplished at the hepatic hilum.
Copyright © 1995 by W.B. Saunders Company INDEX WORDS: Choledochal cyst; anastomotic stricture; primary ductal stricture; cholangitis.
XCISIONAL procedure for choledochal cyst is
now recognised as the treatment of choice. E Lesser procedures such as cystoduodenostomy have
been associated with high morbidity rates and with the potential for malignant change in the biliary tree. 1-4 Excision also has the advantage of stopping reflux of pancreatic juice through pancreaticobiliary maljunction, which is so often associated with choledochal cysts.5 However, recurrent ascending cholangitis and intrahepatic stone formation has necessitated reoperation in 10 of our patients who had previously undergone cyst excision. This article reviews the late complications of excisional surgery and compares the methods of bilioenteric anastomoses. MATERIALS AND METHODS Choledochal cyst excision was performed in 97 patients between February 1969 and April 1994 (Table 1). Reconstruction was performed with either a hepaticoduodenostomy or a Roux-en-Y jejunostomy, and the anastomoses were either below or at the hilum of the hepatic ducts. 1,6Ages ranged from 27 days to 37 years
From the Department of Pediatric Surgery, Kagawa Medical School, Kagawa, Japan. Presented at the 41st Annual International Congress of the British Association of Paediatric Surgeons, Rotterdam, The Netherlands, June 29-July 1, 1994. Address reprint requests to Takuji Todani, MD, Department of Pediatric Surgery, Kagawa Medical School, 1750-1 Miki, Kitagun, Kagawa 761-07, Japan. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3003-0025503. 00/0 478
Table 1. Primary Reconstruction Methods, February 1969 to April 1994 Hepaticoduodenostomy
Hepaticojejujnostomy
At Hilum
At Hilum
Below Hilum
Below Hilum
Type [n]
I [45]
25
5
IV [52] Subtotal
33 58
4 (4) 9 (4)
Total
9
6 (3)
8 (1) 17 (1)
67 (4)
7 (2) 13 (5) 30 (6)
NOTE: I, cystic or diffuse choledochal dilatation; IV, intrahepatic involvement; parentheses indicate number of reoperated patients.
(Table 2), and there was no evidence of malignant change in any of the cysts. Hepaticoenterostomies distal to the hilum were performed in the first 22 patients, and these consisted of hepaticoduodenostomies in 9 cases and hepaticojejunostomies in 13 (Fig 1). One of these cases required reoperation for recurrent cholangitis 7 years after the primary procedure, 6 and 75 subsequent cases were therefore treated with more proximal anastomoses at the level of the hepatic hilum in an attempt to provide a wider anastomosis. In this latter group, hepaticoduodenostomy was the procedure of choice in 58 and Roux-en-Y jejunostomy in 17 cases (Fig 1). RESULTS
Nine of the 22 patients treated with hepaticoenterostomy distal to the hilum required reoperation several years after surgery because of recurrent chop angitis with intrahepatic cholelithiasis secondary to anastomotic stricture formation (Fig 1, Table 3). Five of these patients had undergone primary surgery in our department, and 4 were referred from elsewhere. Late stricturing of hepaticoenteric anastomosis is illustrated by the case of a 9-year-old girl (case 2) who underwent excision for type I cyst and an end-to-side Roux-en-Y hepaticojejunostomy distal to the hilum. The anastomosis measured 16 mm. Episodes of pyrexia and abdominal pain occurred 12 years after surgery. A percutaneous transhepatic cholangiogram Table 2. Age Distribution Age
n
0-12 months 1-5 years 6-10,years 11-20 years 21 years+
25
Total
97
21 17 21 13
Journal of Pediatric Surgery, Vo130, No 3 (March), 1995: pp 478-481
COMPLICATIONS OF CHOLEDOCHAL CYST EXCISION BELOWHILUM
479
AT HILUM
¢Jtt hepaticoduodenostemy
hepatieoj ejunostomy with blind pouch
hepaticoduodenostomy
hepatico jejunostomy end-to-end fashion
13 (5)
22 (9)
"~
~
k
97 (10) ~ Fig 1. Biliary reconstructions performed from February 1969 to April 1994. Numbers in parentheses indicate the number of patients who underwent reoperation.
showed an anastomotic stricture 4 mm in diameter and intrahepatic gallstone formation (Fig 2). Laparotomy showed atrophy of the right lobe of the liver and multiple gallstones. Treatment consisted of right hepatectomy and reanastomosis with an end-to-end hepaticojejunostomy at the hilum. Postoperative progress has been very satisfactory for 3 years. Seventy-four of 75 patients who have undergone hepaticoenterostomy at the hilum with a wide anastomosis have achieved favorable results. The exception was a 17-year-old girl (case 10) who underwent excision for type IV cyst and an end-to-end Rouxen-Y hepaticojejunostomy at the hilum. An intraoperative cholangiogram showed some stricturing of the left hepatic duct near the hilum (Fig 3A). Pyrexia, jaundice, and abdominal pain recurred 15 years later, and a percutaneous transhepatic cholangiogram re-
Fig 2. Case 2. A cholangiogram when patient was 30 years of age taken through PTCD (percutaneous transhepatic cholangio drainage) tube showing anastomotic stricture associated with multiple stones in the hilum and the posterior ducts of the right hepatic lobe,
vealed a severe stricture of the left hepatic duct, possibly a result of a previous incomplete plasty of the hilar duct (Fig 3B). Operation showed no anastomotic stricture but atrophy of the lateral left hepatic
Table 3. Case Summary of Reoperation Age (yr)
Redo-Operation
Age (yr)
Follow-Up(yr)
Anastomotic stricture 1. Hepaticojejunostomy* 2. Hepaticojejunostomy
Type
10 9
15 30
4 3
Hepaticojejunostomy*
16
Hepaticoduodenostomy Hepaticojejunostomy* Hepaticoduodenostomy
11 2 5
23 26 18 6 11
7 2 17 7 6
13 18 15
Hepaticojejunostomy Hepaticojejunostomy and right hepatectomy Hepaticojejunostomy* Hepaticojejunostomy Hepaticojejunostomy Hepaticoduodenostomy Lateral segmentectomy with hepaticojejunostomy Hepaticojejunostomy Hepaticojejunostomy* Hepaticojejunostomy
28 33 24
5 4 3
17
Lateral segmentectomy
32
3
4. IV 5. IV 6. IV
First Operation
7. IV Hepaticojejunostomy 8. IV Hepaticoduodenostomy 9. IV Hepaticoduodenostomy* Primary ductal stricture 10. IV Hepaticojejunostomy *Performed elsewhere.
480
TODANI ET AL
Fig 3. Case 10. (A) An operative cholangiogram at the patient age of 17 years indicating a slight stricture of the left hepatic duct near the hepatic hilum. (B) A cholangiogram when patient was 32 years of age taken through PTC tube showing severe stricture of the left hepatic duct near the hilum with intrahepatic gallstones,
segment. A lateral segmentectomy resulted in a resolution of the symptoms. DISCUSSION
Recurrent symptoms of cholangitis after choledochal cyst excision may be the result of either a stricture at the site of anastomosis or a primary ductal stricture. 6-8 In our series there has been no significant difference in the occurrence rate of cholangitis between hepaticoduodenostomy and Roux-en-Y jejunostomy. 7,8 We have favoured hepaticoduodenostomy because we believe that the operation is more physiological, it is simpler to perform, and there are less complications from adhesive obstruction, anastomotic leakage, and peptic ulceration. 6 Cholangitis secondary to anastomotic stricture has been more frequent in patients who have undergone operations with anastomoses distal to the bifurcation of the hepatic duct and in those with intrahepatic ductal dilatation (type IV cyst). In the group with type IV cysts, anastomotic stricture developed in all four patients with hepaticoduodenostomies performed distal to the hilum and in two of seven of those with jejunostomies anastomosed at similar levels (Table 1). The reasons for this complication may include the
small diameter of the common hepatic duct in many cases of choledochal cyst, a relatively poor blood supply, and epithelial damage and intramural fibrosis. Primary strictures of the hepatic ducts near hilum and intrahepatic ducts are occasionally seen in patients with choledochal cysts, and whenever possible these should be widened by ductoplasties at the time of the primary surgery. Intrahepatic duct strictures may respond to repeated dilatation, although occasionally a segmental hepatic resection may be necessary. A hilar anastomosis allows the construction of a wide anastomosis, which may be performed with a continuous suture, and the chance of anastomotic leakage are lessened. 9 Wide anastomoses are essential to prevent the complication of ascending cholangitis and can be achieved by extending the incisions along the lateral walls of the hepatic ducts 6,1° (Fig 1). This is essential for type IV cysts with intrahepatic involvement. Provided a wide anastomosis was achieved in our series, favorable results could be obtained in both the duodenostomy and the jejunostomy groups. We therefore suggest that surgical enlargement of the hilar ducts to provide wide stoma should be routine in all patients undergoing choledochal cyst excision.
REFERENCES 1. Todani T, Watanabe Y, Narusue M, et al: Congenital bile duct cyst; its classification, operative procedures, and review of 37 cases including cancer arising from choledochal cyst. Am J Surg 134:263-269, 1977 2. Lilly JR: The surgical treatment of choledochal cyst. Surg Gynecol Obstet 149:36-42, 1979
3. Deziel DJ, Rossi RL, Munson L, et al: Management of bile duct cysts in adults. Arch Surg 121:410-415, 1986 4. Yoshikawa K, Yoshida K, Shirai Y, et al: A case of carcinoma arising in the intrapancreatic terminal choledochus 12 years after primary excision of a giant choledochal cyst. Am J Gastroenterol 81:378-384, 1986
COMPLICATIONS OF CHOLEDOCHAL CYST EXCISION
5. Todani T, Watanabe Y, Fujii T, et al: Anomalous arrangement of the pancreatobiliary ductal system in patients with a choledochal cyst. Am J Surg 147:672-676, 1984 6. Todani T, Narusue M, Watanabe Y, et al: Management of congenital choledochal cyst with intrahepatic involvement. Ann Surg 187:272-280, 1978 7. Todani T, Watanabe Y, Toki A, et al: Reoperation for congenital choledochal cyst. Ann Surg 207:142-147, 1988
481
8. Lilly JR: Surgery of coexisting biliary malformations in choledochal cyst. J Pediatr Surg 14:643-647, 1979 9. Terblanche J, Worthley CS, Spence RAJ, et al: High or low hepatico-jejunostomy for bile duct stricture? 108:828-834, 1990 10. Todani T, Watanabe Y, Mizuguchi T, et al: Hepaticoduodenostomy at the hepatic hilum after excision of choledochal cyst. Am J Surg 142:584-581, 1981