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HPB 2003 Volume 5, Number 4 261±263 provided by Elsevier - Publisher Connector DOI 10.1080/13651820310001388 Case report A case of cholecystohepatic with atrophic

A Scho®eld, J Hankins and F Sutherland Department of Surgery, University of Calgary, Calgary, Alberta, Canada

Background Discussion Cholecystohepatic ducts are rare congenital variants of the This case demonstrates a rare congenital anomaly where the biliary tree. ®lls retrograde during an intraoperative cholangio- gram despite clipping of the . The major path of Case outline biliary drainage was through a large cholecystoheptic duct An 81-year-old woman presented with biliary colic and ele- similar to a gallbladder interposition; however, the common vated function tests. An ERCP demonstrated a common hepatic duct was still present but atrophic. This anomaly has stone and stricture of the common hepatic duct. An not been described previously. operative cholangiogram demonstrated an atrophic common hepatic duct and retrograde ®lling of the gallbladder through a Keywords large cholecystoheptic duct. The patient had a cholecystect- cholecystohepatic duct omy and reconstructive cholecystohepatic duct jejunostomy.

Introduction where she underwent an endoscopic cholangiogram Anatomical variation of the biliary tree occurs with (ERCP) and sphincterotomy with successful extraction suf®cient frequency to be of concern to the surgeon of stones. The ERCP also showed that operating in this area. Several persistent fetal connec- there was narrowing of the common hepatic duct,and an tions have been described between the liver and either attempt to pass a guide wire through this area was the gallbladder or the extrahepatic biliary ducts. These unsuccessful. After ERCP,the patient was pain-free and pericholecystic anomalies include the subvesical ducts of was discharged home. Further investigation of this Luschka,cholecystohepatic ducts and gallbladder inter- `stricture' included a magnetic resonance cholangiogram position. (MRCP),which showed mild intrahepatic biliary dilata- True cholecystohepatic connections are rare. We tion but could not further delineate the biliary anatomy. report a case of a woman with a presumed hepatic duct A computed tomography (CT) scan did not show any stricture who was found on intraoperative cholangiogra- evidence of a mass in this area. phy to have an atrophic common hepatic duct and a large The patient returned for laparoscopic cholecystec- cholecystohepatic duct. The major pathway of biliary tomy,with intraoperative cholangiography planned to drainage was through the gallbladder. investigate further the `stricture' and rule out a chol- angiocarcinoma. After clip occlusion of the cystic duct at the gallbladder neck,the ¯uorocholangiogram ®rst Case report showed a dilated distal common bile duct,then ®lling A healthy 81-year-old woman presented with a 3-week of a tortuous atrophic `common hepatic duct'. The history of intermittent epigastric pain,radiating to both intrahepatic ducts then ®lled,with retrograde opaci®ca- shoulder blades,nausea and vomiting. Liver function tion of the gallbladder through a large cholecystohepatic tests showed an obstructive pattern,and an ultrasound duct (Figure 1). The right anterior and posterior sectoral scan of the biliary tree showed cholelithiasis and chole- ducts had separate connections,one above and one docholithiasis. below the take-off of the atrophic common hepatic duct. The patient was transferred to a tertiary care hospital The patient was converted to an open operation.

Correspondence to: Dr F Sutherland, Peter Lougheed Hospital, 3500–26  2003 Taylor & Francis Ave NW, Calgary, Alberta T1Y 6J4, Canada (e-mail: [email protected]) 261 A Schofield et al.

hepatic duct. The extrahepatic biliary tree is initially occluded with endodermal cells,but is later recanalized [4,5]. The anomaly we present,that of cholecystohepatic duct,is thought to arise when there is a persistent connection between the fetal hepatic ducts and the gallbladder,with failure of complete recanalization of the common hepatic duct resulting in its atresia [4] or,in our case,a small atrophic duct. The existence and incidence of `pericholecystic' bile ducts is controversial,and the nomenclature used to describe variations in this area is confusing and inconsistent in the literature [6]. It is common for separate bile ducts to drain variable portions of the right liver and to join the main . Speci®cally,the right anterior and posterior sectors often have separate ducts [7],as was the case in our patient. These mostly represent normal variations [8] and have a reported incidence of 15–20% [9]. Figure 1. Fluoroscopic cholangiography through the proximal cystic duct demon- These normal variations are not to be confused with strates a dilated common bile duct with sequential ®lling of a small tortuous common hepatic duct (arrow) and then the intrahepatic biliary tree; a large cholecystohepatic the pericholecystic bile duct variations discussed below. duct then ®lls the gallbladder behind the occluding clip. These include subvesical ducts (of Luschka) running in the gallbladder plate but not draining into the gallblad- During the removal of the gallbladder a 1-cm diameter der; cholecystohepatic ducts,draining variable portions of bile duct was found to enter the gallbladder approxi- the right lobe directly into the gallbladder lumen,and mately 2 cm above the cystic duct. It was decided to lastly, gallbladder interposition. In 1863 Luschka [10] ®rst reconstruct this large cholecystohepatic duct,as this was described small bile ducts lying in the gallbladder fossa, the major pathway for biliary drainage; a Roux-en-Y either as blind ducts emerging from the right lobe of the hepaticojejunostomy to this duct was performed. Post- liver,or ducts running subserosally to join one of the operatively the patient made a satisfactory recovery,with main biliary channels later. He stated that he did not no complications and speci®cally no postoperative bile believe that these ducts entered the gallbladder lumen, leakage. thereby differing from true cholecystohepatic ducts [11]. Most authors describe these subvesical ducts as 1–2mm Discussion in diameter,draining a variably sized subsegment of the Anatomic variability of the biliary tree is well described right posterior sector and entering the right hepatic duct, [1,2]. Several anatomic studies have described accessory the common hepatic duct or rarely the cystic duct. Their bile ducts as persistent fetal connections between the incidence has ranged in several studies from 12% to 50% liver and the gallbladder or the extrahepatic bile duct [12]. In support of Luschka's theory,Michels [13] system,with an overall incidence quoted as 9 –28% [3]. dissected 500 cadavers and failed to ®nd one case of a These accessory ducts are thought to arise as a result of subvesical duct communicating with the gallbladder disordered biliary tract embryology. During the fourth lumen,as did Healy and Schroy in their corrosion cast week of gestation the hepatic diverticulum arises study [14]. McQuillan and co-workers [15] found that ventrally from the distal foregut and divides into two the subvesical duct most commonly lay in the centre of parts. The hepatic primordium develops cranially,form- the gallbladder bed; the other frequent site was the ing interlacing cords of and the intrahepatic lateral peritoneal re¯ection. Of note is that in none of biliary tree. The gallbladder and cystic duct develop their post-mortem studies was the duct visible before caudally,with a con¯uence of fetal hepatic ducts opening dissection of the gallbladder from its bed. into the proximal cystic duct,to form the common Cystohepatic ducts are de®ned as those draining a

262 Cholecystohepatic duct with atrophic common hepatic duct variable portion of the right lobe into the cystic duct. reconstruction with a Roux-en-Y hepaticojejunostomy, Some authors use this term to include connections to the as in this case. gallbladder (cholecystohepatic ducts) [16]. True chole- cystohepatic ducts are rare [2 4,6,17]. Several studies – References have shown combined incidences of cystohepatic and cholecystohepatic ducts of between 0.2% and 2.3% [16]. 1 Flint ER. Abnormalities of the right hepatic,cystic and A large series based on operative and cholangiographic gastroduodenal arteries and of the bile ducts. Br J Surg 1923;10:509. ®ndings demonstrated that the overall incidence of cysto- 2 Izzidien AY. Cholecystohepatic duct. J R Coll Surg Edinb hepatic and cholecystohepatic ducts was 0.85% (12 cases 1979;24:361–2. in 1410 ). However,only one of the 12 3 Klotz HP,Schlumpf R,Largiader F. Injury to an accessory cases was an actual cholecystohepatic duct [16]. bile duct during laparoscopic . Surg Cystohepatic and cholecystohepatic ducts may occur Laparosc Endosc 1992;2:317–20. 4 Kihne MJ, et al. Persistent cholecystohepatic ducts. Arch in the presence of normal common hepatic and common Surg 1980;115:972–4. bile ducts [2]. As noted above,the hepatic territory 5 Moore KL. The Developing Human – Clinically Orientated drained by these ducts is variable. In the series of Cham- Embryology,3rd edn. Toronto: Mosby,1982. petier and colleagues [16] the majority were segmental or 6 Javors BR,Simmons MZ,Wachsberg RH. Cholangio- subsegmental ducts. Two of the cystohepatic ducts acted graphic demonstration of the cholecystohepatic duct of Luschka. Abdomin Imaging 1998;23:620–1. as right hepatic ducts,draining segments 5,6,7 and 8 7 Couinaud C. Le Foie: Etudes Anatomiques et Chirurgicales. into the cystic duct. Drainage of the right,left,or both, Paris: Masson,1957. hepatic ducts into the gallbladder is infrequent [2,18]. 8 Skandalakis JE,Gray SW,Rowe JS. Anatomical Complica- Complete interposition of the gallbladder has been tions in General Surgery. McGraw-Hill,1983. 9 Thompson RW,Schuler JG. Bile peritonitis from a chole- reported in a few individuals [17,19]. In these cases cystohepatic bile ductule: an unusual complication of both the right and left hepatic ducts drain directly into cholecystectomy. Surgery 1986;99:511–13. the gallbladder,either separately or as one main duct, 10 Luschka H. Die Anatomie des Menschlichen Bauches. with associated absence of the common hepatic and Tubingen: H Lauppschen,Buchhandlung 1863;255. cystic duct. The sole pathway for biliary drainage is 11 Jamshidi M,Obermeyer RJ,Garcia G,Hashmi M. Post- laparoscopic cholecystectomy bile leak secondary to an therefore via the gallbladder. accessory duct of Luschka. Int Surg 1999;84:86–8. We believe our case to be unique,in that it almost 12 Gupta SC,Gupta CD,Arora AK,Gupta SB. A study of represents gallbladder interposition; the large cholecys- subvesical duct in corrosion casts. Indian J Med Res 1977; tohepatic duct was the major drainage conduit for the 66:338–40. 13 Michels NA. Blood Supply and Anatomy of the Upper right and left hepatic ducts. However,in addition our Abdominal Organs with a Descriptive Atlas. Philadelphia: JB patient had a narrow,tortuous common hepatic duct Lippincott,1995. remnant. Kihne et al. [4] reported a similar,but not 14 Healey JE,Schroy PC. Anatomy of the biliary ducts within identical,case in which a cholecystohepatic duct drained the human liver. Analysis of the prevailing pattern of the intrahepatic junction of the right and left hepatic branching and the major variations of the biliary ducts. Arch Surg 1953;66:599–616. ducts through the gallbladder and through a small 15 McQuillan T,Manolas SG,Hayman JA,Kune KA. Surgi- stenosed cystic duct into the distal common duct. cal signi®cance of the bile duct of Luschka. Br J Surg 1989; The importance of these variations in biliary anatomy 76:696–8. lies in their vulnerable position during cholecystectomy 16 Champetier J,Letoublon C,Alnaason I,Charvin B. The [3]. Only occasionally may they be seen by preoperative cystohepatic ducts: surgical implications. Surg Radiol Anat 1991;13:203–11. endoscopic cholangiography or MRCP. Most of these 17 Braasch JW. Congenital anomalies of the gallbladder and ducts are too small to be delineated [6],and even a large bile ducts. Surg Clin North Am 1958;38:627–30. anatomic variation may not be appreciated,as in our 18 Stokes TL,Old L. Cholecystohepatic duct. Am J Surg case. Therefore,these anomalies are rarely identi®ed 1978;135:703–5. 19 Stringer DA,Dobranowski J,Ein SH,Roberts EA,Dane- before operation. Accidental damage to a duct draining man A,Filler RM. Interposition of the gallbladder – or the an extensive hepatic territory should be repaired im- absent common hepatic duct and cystic duct. Pediatr Radiol mediately,by either direct suturing over a T-tube or 1987;17:151–3.

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