J Ped Surg Case Reports 3 (2015) 257e259
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Percutaneous transhepatic balloon dilatation of the hepaticojejunal anastomosis for the definitive treatment of the intrahepatic bile duct stones occurring in choledochal cysts excised children uz Ates¸ a, Süleyman Men b, Osman Z. Karakus¸ a, *, Gülce Hakgüder a, Aytaç Gülcü b, Og a a Mustafa Olguner , Feza M. Akgür a b
_ Department of Pediatric Surgery, Dokuz Eylül University, Medical School, Izmir, Turkey _ Department of Radiology, Dokuz Eylül University, Medical School, Izmir, Turkey
a r t i c l e i n f o
a b s t r a c t
Article history: Received 8 December 2014 Received in revised form 27 April 2015 Accepted 28 April 2015
Cyst excision with hepaticojejunostomy is the well defined standard treatment for choledochal cysts. Intra or extrahepatic bile duct stone formation are serious complications observed in long-term followup after hepaticojejunostomy. Revision of hepaticojejunostomy is the most defined treatment of intra or extrahepatic bile duct stones secondary to hepaticojejunal anastomotic stenosis. We report two cases of common hepatic duct and/or intrahepatic bile duct stones that developed after hepatico-jejunostomy for choledochal cyst resection, that were treated with percutaneous transhepatic balloon dilatation of the hepaticojejunal anastomotic stenosis. During percutaneous transhepatic cholangiography a catheter was passed through the hepaticojejunal anastomotic stenosis and the hepatic duct-jejunal anastomotic junction was dilated with a balloon. The common hepatic duct stone and intrahepatic bile duct stones thus passed to the jejunum following the dilatation. The patients recovered uneventfully and are free of stones. Ó 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Key words: Hepaticojejunostomy Percutaneous transhepatic cholangiography Intrahepatic bile duct stones Extrahepatic bile duct stones Choledochal cyst
Cyst excision with hepaticojejunostomy is the standard treatment for choledochal cysts [1,2]. One of the late complications of hepaticojejunostomy is intra or extrahepatic bile duct stone formation secondary to hepaticojejunal anastomotic stenosis [1e5]. Various treatment methods such as revision of the hepaticojejunostomy, intracorporeal electrohydraulic lithotripsy, extracorporeal shockwave lithotripsy and even segmental hepatectomy have been performed for the treatment of intra or extrahepatic bile duct stones [5e7]. We herein report two cases of common hepatic duct and/or intrahepatic bile duct stones secondary to hepaticojejunal anastomotic stenosis that were treated with percutaneous transhepatic balloon dilatation of the hepaticojejunal anastomotic stenosis.
* Corresponding author. Dokuz Eylül Üniversitesi, Tıp Fakültesi, Çocuk Cerrahisi _ Anabilim Dalı, 35340 Balçova, Izmir, Turkey. Tel.: þ90 232 412 30 02; fax: þ90 232 279 21 01. E-mail address:
[email protected] (O.Z. Karakus¸).
1. Case reports 1.1. Case #1 A 26-year-old man, operated for Type 1 choledochal cyst at the age of 6, presented with recurrent cholangitis. Computerized tomography (CT) showed dilatation of the bile ducts proximal to the hepaticojejunal anastomosis. Common hepatic duct stone and multiple intrahepatic bile duct stones were present in the left hepatic segments. Percutaneous transhepatic cholangiography (PTC) showed left sided intrahepatic bile duct stones, a large cone shaped common hepatic duct stone (5 cm long) and hepaticojejunal anastomotic stenosis (Fig. 1). During PTC a guide was introduced from the right side of the main hepatic duct stone and the stenotic hepaticojejunal anastomosis was dilated with a balloon (Boston Scientific, Natick, USA) (Fig. 2). Thus, the common hepatic duct stone passed through hepaticojejunal anastomosis to the jejunum following the dilatation procedure. Afterward all intrahepatic stones passed through from the dilated hepaticojejunal anastomosis into the jejunum. The trans-anastomotic 8 fr external biliary drainage catheter (Flexima,
2213-5766/Ó 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). http://dx.doi.org/10.1016/j.epsc.2015.04.015
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Fig. 1. PTC showing multiple left intrahepatic bile duct stones, a large cone shaped common hepatic duct stone and hepaticojejunal anastomotic stenosis (arrow).
Boston Scientific, Natick, USA) was left to ensure bile flow through the dilated hepaticojejunal anastomosis after the PTC. PTC was repeated a week later and hepaticojejunal anastomosis was found to be patent and the catheter was removed. Repeated CT showed clearance of all intrahepatic bile duct stones. The patient recovered uneventfully and is free of stones after 27 months follow-up without any symptoms.
1.2. Case #2 A 15-year-old girl operated for Type 1 choledochal cyst when she was 10 years old. She presented with recurrent cholangitis. CT showed dilation of the bile ducts proximal to the hepaticojejunostomy anastomosis and multiple intrahepatic bile duct stones.
Fig. 2. PTC showing passage of guide from the right side of common hepatic duct stone.
Fig. 3. PTC showing multiple left intrahepatic bile duct stones and hepaticojejunal anastomotic stenosis (arrow).
PTC showed multiple left sided intrahepatic bile duct stones and hepaticojejunal anastomotic stenosis (Fig. 3). During PTC, a guide was passed through the hepaticojejunal anastomotic stenosis and the hepaticojejunal anastomosis was dilated with a balloon (Boston Scientific, Natick, USA) (Fig. 4). Following dilation, all of the intrahepatic stones passed through the hepaticojejunal anastomosis into the jejunum. The trans-anastomotic 8 fr external biliary drainage catheter (Flexima, Boston Scientific, Natick, USA) was left to ensure bile flow through the dilated hepaticojejunal anastomosis after the PTC. PTC was repeated a week later and hepaticojejunal anastomosis was found to be patent and catheter was removed. Repeated CT showed clearance of all intrahepatic bile duct stones. The patient
Fig. 4. PTC showing passage of guide from bilioenteric anastomotic stenosis.
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recovered uneventfully and is free of stones after 23 months followup without any symptoms. 2. Discussion Cyst excision with Roux-en-Y hepaticojejunostomy is the most preferred treatment for choledochal cysts [1,2]. Intra or extrahepatic bile duct stones formation are the long term serious complications of choledochal cyst treatment. Intrahepatic bile stone formation has been shown to occur mostly in patients operated older than 5 years [4,5]. Biliary stasis secondary to the hepaticojejunostomy anastomosis stenosis or residual dilated intrahepatic bile ducts are considered to be the causes of intrahepatic bile stone formation [1e5]. Bile statis can cause biliary infection and recurrent cholangitis, thus facilitating stone formation. The presence of ascendant gram negative enterobacteria in the intrahepatic biliary tree has been shown to play an important role in intrahepatic bile stone formation [8]. Bile sludge present in the intrahepatic bile ducts has also been shown to contribute to intrahepatic bile stone formation [9,10]. Intraoperative endoscopy and irrigation of the biliary system during choledochal cyst excision have been recommended to prevent intrahepatic bile stone formation [10,11]. Excision of the extrahepatic bile duct starting from the confluence of the hepatic duct till pancreaticobiliary junction, together with wide hepaticojejunostomy is recommended for the prevention of cholangitis and intrahepatic bile stone formation [12]. Revision of the hepaticojejunostomy is the most utilized treatment of intrahepatic bile stones secondary to hepaticojejunal anastomotic stenosis [5,12,13]. Intracorporeal electrohydraulic lithotripsy has been utilized for biliary stones with normal hepaticojejunal anastomosis. Flexible cholangioscope is introduced through jejunal limb during laparotomy for this purpose; the stones are fragmented with electrohydraulic lithotripsy and irrigated [6]. In the presence of a septal intrahepatic bile duct stenosis, the stenotic septa can be resected with a grasper introduced through the choloangioscope and stones can be removed [14]. Minimally invasive methods such as percutaneous transhepatic cholangioscopic lithotripsy (PTCL) and extracorporeal shockwave lithotripsy (ESWL) have been described for the treatment of intrahepatic bile duct stones with normal hepaticojejunal anastomosis [4,7,12]. Multiple sessions of ESWL is needed for a successful lithotripsy [7]. Balloon dilatation of intrahepatic bile duct septa has been reported in conjunction to PTCL in two cases of bile duct stenosis accompanying intrahepatic stones [12]. To the best of our knowledge percutaneous transhepatic balloon dilatation of the hepaticojejunal anastomotic stenosis to treat common hepatic duct and/or intrahepatic bile duct stones secondary to hepaticojejunal anastomotic stenosis has not been reported. In the present cases
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balloon dilatation of the hepaticojejunal anastomotic stenosis was sufficient to discharge the stones together with the accumulated bile. No lithotripsy procedure was performed. Even the 5 cm long stone passed through the hepaticojejunal anastomosis to jejunum after dilatation. 3. Conclusion Balloon dilatation of the hepaticojejunal anastomotic stenosis during PTC is a minimally invasive, safe and effective method in the treatment of intra and/or extrahepatic bile duct stones formed after choledochal cyst excision. References [1] Chijiiwa K, Koga A. Surgical management and long-term follow-up of patients with choledochal cysts. Am J Surg 1993;165(2):238e42. [2] Ono S, Fumino S, Shimadera S, Iwai N. Long-term outcomes after hepaticojejunostomy for choledochal cysts: a 10-to-27-year follow-up. J Pediatr Surg 2010;45:376e8. [3] Chijiiwa K, Tanaka M. Late complications after excisional operation in patients with choledochal cyst. J Am Coll Surg 1994;179:139e44. [4] Miyano T, Yamataka A, Kato Y, Segawa O, Lane G, Takamizawa S, et al. Hepaticoenterostomy after excision of choledochal cysts in children: a 30 years experience with 180 cases. J Pediatr Surg 1996;31:1417e21. [5] Yamataka A, Ohshiro K, Okada Y, Hosoda Y, Fujiwara T, Kohno S, et al. Complications after cyst excision with hepaticoenterostomy for choledochal cysts and surgical management in children versus adults. J Pediatr Surg 1997;32: 1097e102. [6] Shima H, Yamataka A, Yanai T, Kobayashi H, Miyano T. Intracorporeal electrohydraulic lithotripsy for intrahepatic bile duct stone formation after choledochal cyst excision. Pediatr Surg Int 2004;20:70e2. [7] Okada Y, Miyamoto M, Yamazaki T, Motoi I, Kuribayashi M, Kodoma K. Piezzoelectric extracorporeal shockwave lithotripsy for bile duct stone formation after choledochal cyst excision. Pediatr Surg Int 2007;23:357e60. [8] Kaneko K, Ando H, Seo T, Ono Y, Ochiai K, Ogura Y. Bile infection contributes to intrahepatic calculi formation after excision of choledochal cysts. Pediatr Surg Int 2005;21:8e11. [9] Shimotakahara A, Yamataka A, Kobayashi H, Yanai T, Lane GJ, Miyano T. Massive debris in the intrahepatic bile ducts in choledochal cyst: possible cause of postoperative stone formation. Pediatr Surg Int 2004;20:67e9. [10] Takahashi T, Shimotakahara A, Okazaki T, Koga H, Miyano G, Lane HJ, et al. Intraoperative endoscopy during choledochal cyst excision; extended longterm follow-up compared with recent cases. J Pediatr Surg 2010;45:379e82. [11] Miyano T, Yamataka A, Kato Y, Kohno S, Fujiwara T. Choledochal cysts:special emphasis on the usefulness of intraoperative endoscopy. J Pediatr Surg 1995; 30:482e4. [12] Urushihara N, Fukumato K, Fukuzawa H, Mitsunaga M, Watanabe K, Aoba T, et al. Long-term outcomes after excision of choledochal cysts in a single institution: operative procedures and late complications. J Pediatr Surg 2012; 47:2169e74. [13] Ando H, Kaneko K, Ito F, Sea T, Ito T. Operative treatment of congenital stenoses of the intrahepatic bile ducts in patients with choledochal cysts. Am J Surg 1997;173:491e4. [14] Ono Y, Kaneko K, Ogura Y, Sumida W, Tainaka T, Seo T, et al. Endoscopic resection of intrahepatic septal stenosis: minimally invasive approach to manage hepatolithiasis after choledochal cyst excision. Pediatr Surg Int 2006; 22:939e41.
