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Traumatic rupture of choledochal cyst in a child
Journal of Pediatric Surgery (2005) 40, E7 – E8
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Traumatic rupture of choledochal cyst in a child Itsuro Nagae*, Akihiko Tsuchida, Yoshihide Tanabe, Soshi Takahashi, Shintaro Minato, Yasuhisa Koyanagi, Tatsuya Aoki Third Department of Surgery, Tokyo Medical University, Shinjuku-ku,Tokyo 160-0023, Japan Index words: Choledochal cyst; Traumatic rupture; Pancreaticobiliary maljunction
Abstract Traumatic rupture of choledochal cyst is an extremely rare disorder. The current patient is a 4year-old boy who fell in a bathroom and suffered a blow to the abdomen. Percutaneous transhepatic cholangiography revealed pancreaticobiliary maljunction. Inflammation of the peritoneal cavity was moderate. At first look, the choledochal cyst was excised and hepaticojejunostomy was performed. At this time, a rupture approximately 2 mm in diameter was recognized at the rear surface of the inferior part of the common bile duct. D 2005 Elsevier Inc. All rights reserved.
Choledochal cyst is a rare congenital disorder, and in nearly all cases, it is accompanied by pancreaticobiliary maljunction (PBM) [1]. In rare instances, choledochal cyst is complicated by spontaneous perforation [2], but reports of traumatic rupture are exceedingly sparse [3-8]. We report on a case of traumatic rupture of choledochal cyst.
1. Case report A 4-year-old boy fell in a bathroom and suffered a blow to the abdomen. At the time of the injury, the boy experienced mild abdominal pain, but this gradually intensified; fever developed and the boy was admitted to hospital 5 days later. Abdominal computed tomography revealed ascites and a normal-sized gallbladder, swelling of the intrahepatic bile duct, and distension of the common bile duct (Fig. 1). After perforation to the abdominal cavity under echo guide, the aspirate was dark yellow bilious
* Corresponding author. Tel.: +81 3 3342 6111x5080; fax: +81 3 3340 4575. E-mail address: [email protected] (I. Nagae). 1531-5037/05/4002-0032$30.00/0 D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2004.10.007
ascites, which contained 2904 U/L amylase, and total bilirubin was 10.05 mg/dL. In light of these findings, peritonitis owing to rupture of a choledochal cyst was diagnosed and emergency surgery was performed. Initially, percutaneous transhepatic cholangiodrainage (PTCD) was established, and then cholangiography revealed the presence of PBM and cystic dilation in the common bile duct (Fig. 2). Next, laparoscopic examination of the abdominal cavity disclosed that intraabdominal inflammation was moderate. Accordingly, laparotomy and cholecystectomy were performed, followed by resection of the choledochal cyst and hepaticojejunostomy. During surgery, a rupture approximately 2 mm in diameter was confirmed at the rear surface of the inferior part of the common bile duct. Postoperative convalescence was uneventful, and the patient was discharged on the 17th day of hospitalization.
2. Discussion Choledochal cyst with PBM is a congenital anomaly. In some cases of PBM, there is no dilation of the common bile duct. Yet it is believed that whether there is dilation depends on the degree of development of elastic fiber in the bile duct
E8 wall during the neonatal period [2,6]. In general, elastic fiber in the human bile duct wall during the neonate period has not been verified, but its appearance is observed 6 months after birth. Cases of choledochal cyst have been recognized prenatally [9], yet whether there is dilation of the common bile duct owing to congenital or acquired factors is still obscure. In choledochal cyst, development of the common bile duct wall is weak, and spontaneous perforation is observed in 1.8% to 7% of all cases [2]. By comparison, the incidence of bile duct rupture caused by regular blunt abdominal injury other than to the choledochal cyst is 0.4% to 0.8% [10], so it is conjectured that trauma makes it even easier for perforations to occur. Nevertheless, reports of traumatic rupture of choledochal cyst are extremely rare. As far as we know, there have only been 7 such reports including the present one [3-8]. Currently, in the treatment of rupture of choledochal cyst, abdominal and bile duct drainage is established, and once inflammation begins, a second look operation is recommended [8]. However, if systemic conditions are stable and if peritonitis is moderate, first look cholecystectomy, resection of the choledochal cyst, and hepaticojejunostomy should be performed. In nearly all cases of choledochal cyst, there is also PBM, and in the future, biliary tract cancer will be involved at a high rate [11]. For this reason, preventive diversion operation, in which retentive bile duct is reduced as much as possible, is required. In general, bile is aseptic, and even if bile drains into the abdominal cavity from a bile duct perforation, the possibility of this condition developing into generalized peritonitis is slight because such peritonitis will be chemical peritonitis caused by bile or by pancreatic juice mixed in the bile. There was a certain delay in making a diagnosis and starting treatment [2]. Given this situation, there have been many reports in which a definitive diagnosis was obtained by verifying the presence of bilious ascites from intraperitoneal perforation. In recent years, however, with progress in imaging diagnosis, it has become possible to diagnose the condition early. It is feared that second look operation is encouraged excessively and that the optimal operation is not selected even for cases in which first look radical surgery would be possible. It is the opinion of the authors that as progress is made in diagnostic method, thinking regarding treatment methods must also change. In our case, the reason
Fig. 1 Abdominal computed tomography shows a normal-sized gallbladder, swelling of the intrahepatic bile duct, and distension of the common bile duct.
I. Nagae et al.
Fig. 2 Intraoperative cholangiography via PTCD shows PBM with perforation of a posterior wall of the lower common bile duct (dark arrow).
that PTCD and abdominal laparoscopy were performed during surgery is that after bile duct drainage, the degree of peritonitis was assessed, and if inflammations were marked, abdominal drainage would be established and the operation would be completed. In determining the suitability of surgery for traumatic rupture of the bile duct, laparoscopy is extremely helpful. In view of the progress that has been made in surgery and in imaging diagnostics, cases in which radical surgery is performed at first look can be expected to increase in the future.
References [1] Komi N, Takehara H, Kunitomo K. Choledochal cyst: anomalous arrangement of the pancreaticobiliary ductal system and biliary malignancy. J Gastroenterol Hepatol 1989;4:63 - 74. [2] Ando K, Miyano T, Kohno S, et al. Spontaneous perforation of choledochal cyst: a study of 13 cases. Eur J Pediatr Surg 1998;8:23 - 5. [3] Ellis H, Cronin K. Bile peritonitis. Br J Surg 1961;48:166 - 71. [4] Chen WJ, Chang CH, Hung WT. Congenital choledochal cyst: with observations on rupture of the cyst and intrahepatic ductal dilatation. J Pediatr Surg 1973;8:529 - 38. [5] Battersby C. Ruptured choledochal cyst: recognition and management. Aust N Z J Surg 1978;5:515 - 7. [6] Nimura H, Murai R, Watanabe K, et al. A case of traumatic rupture of choledochal cyst in infant. Fukubu Kyukyu Shinryo no Shinpo 1989; 9:1033 - 6. [7] Sandor L, Bali I, Bozo A, et al. Rupturierte retroperitoneale gallengangszyste. Unfallchirurg 1991;94:360 - 2. [8] Raj JP, Walsh M. Choledochal cyst and blunt trauma—case report. Injury 2002;33:644 - 6. [9] MacKenzie TC, Howell LJ, Flake AW, et al. The management of prenatally diagnosed choledochal cyst. J Pediatr Surg 2001;36:1241 - 3. [10] Pachter HL, Liang HG, Hofstetter SR. Liver and biliary tract trauma. In: Felliciano DV, Moore EE, Kenneth LM, editors. Trauma. Stanford7 Appleton & Lange; 1996. p. 487 - 523. [11] Tashiro S, Imaizumi T, Ohkawa H, et al. Pancreaticobiliary maljunction: retrospective and nationwide survey in Japan. J Hepatobiliary Pancreat Surg 2003;10:345 - 51.
www.elsevier.com/locate/jpedsurg
Traumatic rupture of choledochal cyst in a child Itsuro Nagae*, Akihiko Tsuchida, Yoshihide Tanabe, Soshi Takahashi, Shintaro Minato, Yasuhisa Koyanagi, Tatsuya Aoki Third Department of Surgery, Tokyo Medical University, Shinjuku-ku,Tokyo 160-0023, Japan Index words: Choledochal cyst; Traumatic rupture; Pancreaticobiliary maljunction
Abstract Traumatic rupture of choledochal cyst is an extremely rare disorder. The current patient is a 4year-old boy who fell in a bathroom and suffered a blow to the abdomen. Percutaneous transhepatic cholangiography revealed pancreaticobiliary maljunction. Inflammation of the peritoneal cavity was moderate. At first look, the choledochal cyst was excised and hepaticojejunostomy was performed. At this time, a rupture approximately 2 mm in diameter was recognized at the rear surface of the inferior part of the common bile duct. D 2005 Elsevier Inc. All rights reserved.
Choledochal cyst is a rare congenital disorder, and in nearly all cases, it is accompanied by pancreaticobiliary maljunction (PBM) [1]. In rare instances, choledochal cyst is complicated by spontaneous perforation [2], but reports of traumatic rupture are exceedingly sparse [3-8]. We report on a case of traumatic rupture of choledochal cyst.
1. Case report A 4-year-old boy fell in a bathroom and suffered a blow to the abdomen. At the time of the injury, the boy experienced mild abdominal pain, but this gradually intensified; fever developed and the boy was admitted to hospital 5 days later. Abdominal computed tomography revealed ascites and a normal-sized gallbladder, swelling of the intrahepatic bile duct, and distension of the common bile duct (Fig. 1). After perforation to the abdominal cavity under echo guide, the aspirate was dark yellow bilious
* Corresponding author. Tel.: +81 3 3342 6111x5080; fax: +81 3 3340 4575. E-mail address: [email protected] (I. Nagae). 1531-5037/05/4002-0032$30.00/0 D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2004.10.007
ascites, which contained 2904 U/L amylase, and total bilirubin was 10.05 mg/dL. In light of these findings, peritonitis owing to rupture of a choledochal cyst was diagnosed and emergency surgery was performed. Initially, percutaneous transhepatic cholangiodrainage (PTCD) was established, and then cholangiography revealed the presence of PBM and cystic dilation in the common bile duct (Fig. 2). Next, laparoscopic examination of the abdominal cavity disclosed that intraabdominal inflammation was moderate. Accordingly, laparotomy and cholecystectomy were performed, followed by resection of the choledochal cyst and hepaticojejunostomy. During surgery, a rupture approximately 2 mm in diameter was confirmed at the rear surface of the inferior part of the common bile duct. Postoperative convalescence was uneventful, and the patient was discharged on the 17th day of hospitalization.
2. Discussion Choledochal cyst with PBM is a congenital anomaly. In some cases of PBM, there is no dilation of the common bile duct. Yet it is believed that whether there is dilation depends on the degree of development of elastic fiber in the bile duct
E8 wall during the neonatal period [2,6]. In general, elastic fiber in the human bile duct wall during the neonate period has not been verified, but its appearance is observed 6 months after birth. Cases of choledochal cyst have been recognized prenatally [9], yet whether there is dilation of the common bile duct owing to congenital or acquired factors is still obscure. In choledochal cyst, development of the common bile duct wall is weak, and spontaneous perforation is observed in 1.8% to 7% of all cases [2]. By comparison, the incidence of bile duct rupture caused by regular blunt abdominal injury other than to the choledochal cyst is 0.4% to 0.8% [10], so it is conjectured that trauma makes it even easier for perforations to occur. Nevertheless, reports of traumatic rupture of choledochal cyst are extremely rare. As far as we know, there have only been 7 such reports including the present one [3-8]. Currently, in the treatment of rupture of choledochal cyst, abdominal and bile duct drainage is established, and once inflammation begins, a second look operation is recommended [8]. However, if systemic conditions are stable and if peritonitis is moderate, first look cholecystectomy, resection of the choledochal cyst, and hepaticojejunostomy should be performed. In nearly all cases of choledochal cyst, there is also PBM, and in the future, biliary tract cancer will be involved at a high rate [11]. For this reason, preventive diversion operation, in which retentive bile duct is reduced as much as possible, is required. In general, bile is aseptic, and even if bile drains into the abdominal cavity from a bile duct perforation, the possibility of this condition developing into generalized peritonitis is slight because such peritonitis will be chemical peritonitis caused by bile or by pancreatic juice mixed in the bile. There was a certain delay in making a diagnosis and starting treatment [2]. Given this situation, there have been many reports in which a definitive diagnosis was obtained by verifying the presence of bilious ascites from intraperitoneal perforation. In recent years, however, with progress in imaging diagnosis, it has become possible to diagnose the condition early. It is feared that second look operation is encouraged excessively and that the optimal operation is not selected even for cases in which first look radical surgery would be possible. It is the opinion of the authors that as progress is made in diagnostic method, thinking regarding treatment methods must also change. In our case, the reason
Fig. 1 Abdominal computed tomography shows a normal-sized gallbladder, swelling of the intrahepatic bile duct, and distension of the common bile duct.
I. Nagae et al.
Fig. 2 Intraoperative cholangiography via PTCD shows PBM with perforation of a posterior wall of the lower common bile duct (dark arrow).
that PTCD and abdominal laparoscopy were performed during surgery is that after bile duct drainage, the degree of peritonitis was assessed, and if inflammations were marked, abdominal drainage would be established and the operation would be completed. In determining the suitability of surgery for traumatic rupture of the bile duct, laparoscopy is extremely helpful. In view of the progress that has been made in surgery and in imaging diagnostics, cases in which radical surgery is performed at first look can be expected to increase in the future.
References [1] Komi N, Takehara H, Kunitomo K. Choledochal cyst: anomalous arrangement of the pancreaticobiliary ductal system and biliary malignancy. J Gastroenterol Hepatol 1989;4:63 - 74. [2] Ando K, Miyano T, Kohno S, et al. Spontaneous perforation of choledochal cyst: a study of 13 cases. Eur J Pediatr Surg 1998;8:23 - 5. [3] Ellis H, Cronin K. Bile peritonitis. Br J Surg 1961;48:166 - 71. [4] Chen WJ, Chang CH, Hung WT. Congenital choledochal cyst: with observations on rupture of the cyst and intrahepatic ductal dilatation. J Pediatr Surg 1973;8:529 - 38. [5] Battersby C. Ruptured choledochal cyst: recognition and management. Aust N Z J Surg 1978;5:515 - 7. [6] Nimura H, Murai R, Watanabe K, et al. A case of traumatic rupture of choledochal cyst in infant. Fukubu Kyukyu Shinryo no Shinpo 1989; 9:1033 - 6. [7] Sandor L, Bali I, Bozo A, et al. Rupturierte retroperitoneale gallengangszyste. Unfallchirurg 1991;94:360 - 2. [8] Raj JP, Walsh M. Choledochal cyst and blunt trauma—case report. Injury 2002;33:644 - 6. [9] MacKenzie TC, Howell LJ, Flake AW, et al. The management of prenatally diagnosed choledochal cyst. J Pediatr Surg 2001;36:1241 - 3. [10] Pachter HL, Liang HG, Hofstetter SR. Liver and biliary tract trauma. In: Felliciano DV, Moore EE, Kenneth LM, editors. Trauma. Stanford7 Appleton & Lange; 1996. p. 487 - 523. [11] Tashiro S, Imaizumi T, Ohkawa H, et al. Pancreaticobiliary maljunction: retrospective and nationwide survey in Japan. J Hepatobiliary Pancreat Surg 2003;10:345 - 51.