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cbd duct

The common bile duct (CBD) was measured in 600 ultrasound studies. In normal patients the mean width of the CBD increased from 2.8 mm in the age group 20 years or younger to 4.1 mm in patients 71 years of age or older. The mean width of the CBD for all age groups in normal cases was 3.4 (range 2 to 11 mm). In presence of cholelithiasis the mean width of the CBD was 4.5 mm (range 2 to 15 mm). There was no further increase of the CBD after cholecystectomy (mean width 4.6 mm, range 2 to 12 mm). The CBD in patients with hepatocellular disease was wider than normals (mean 4.4 mm) and markedly increased in patients with pancreatic disease (mean 6.2 mm, range 2 to 21 mm) due to obstruction in several cases.

Cbd duct

Transabdominal ultrasonography showing four parallel ducts.

In conclusion, a case of DCBD associated with APBJ and choledocholithiasis was reported. In case of DCBD the opening site of ACBD is of greatest clinical importance because of its close implications with concomitant pathology. The adequate diagnosis of this rare anomaly is stressed since the possible operative complications may occur when DCBD is not recognized prior to surgical treatment.

CASE REPORT

It seems that double common bile duct is a very rare anomaly in western world, since Teilum[3] identified only 24 cases in western literature until 1986. On the other hand, Yamashita reviewed Japanese literature from 1968 to 2002 and found 47 patients with this anomaly[4].

Double common bile duct (DCBD) is a rare congenital anomaly in which two common bile ducts exist. One usually has normal drainage into the papilla duodeni major and the other usually named accessory common bile duct (ACBD) opens in different parts of upper gastrointestinal tract (stomach, duodenum, ductus pancreaticus or septum). This anomaly is of great importance since it is often associated with biliary lithiasis, choledochal cyst, anomalous pancreaticobiliary junction (APBJ) and upper gastrointestinal tract malignancies. We recently recognized a rare case of DCBD associated with APBJ with lithiasis in better developed common bile duct. The opening site of ACBD was in the pancreatic duct. The anomaly was suspected by transabdominal ultrasonography and finally confirmed by endoscopic retrograde cholangiopancreatography (ERCP) followed by endoscopic sphincterotomy and stone extraction. According to the literature, the existence of DCBD with the opening of ACBD in the pancreatic duct is most frequently associated with APBJ and gallbladder carcinoma. In case of DCBD, the opening site of ACBD is of greatest clinical importance because of its close implications with concomitant pathology. The adequate diagnosis of this rare anomaly is significant since the operative complications may occur in cases with DCBD which is not recognized prior to surgical treatment.

DISCUSSION

Double common bile duct (DCBD) is a rare congenital anomaly in which two common bile ducts exist. One usually has normal drainage into the duodenum and the other usually named accessory common bile duct (ACBD) opens in different parts of upper gastrointestinal tract (stomach, duodenum, ductus pancreaticus or septum). Bile from different parts of the liver is drained through the ACBD. This anomaly is of great importance since it is often associated with lithiasis in the ectopic bile duct, choledochal cyst, anomalous pancreaticobiliary junction (APBJ) and upper gastrointestinal tract malignancies. Anomalous pancreaticobiliary junction is a congenital anomaly in which the pancreatic and biliary ducts are joined outside the duodenal wall forming a long common channel[1]. It is well known that APBJ is frequently associated with congenital choledochal cyst and cancer of the biliary system regardless of DCBD existence[2].

Cbd duct

The patient remained unstable and required inotropic agents, artificial respirator support and continuous hemodiafiltration in the intensive care unit until postoperative day 10. Furthermore, multiple additional drainage, and administration of antibacterial and antifungal agents were required for the refractory intra-abdominal and retroperitoneal abscesses. On day 16, active bleeding was observed at the abdominal wall around the T-tube, and hemostasis was achieved by transcatheter arterial embolization. On day 55, T-tube cholangiography revealed that the impacting CBD stone had disappeared naturally and, thus, the long common channel of the pancreaticobiliary duct and the diagnosed pancreaticobiliary maljunction could be observed (Fig. 5). After removal of the T-tube on day 136, she was transferred to the hospital for recuperation on day 152.

A 71-year-old woman with consciousness disorder was transported to the emergency department of another hospital. She was diagnosed as having severe peritonitis with septic shock and transferred to our hospital for emergency surgery. Enhanced computed tomography (CT) revealed supraduodenal CBD dilation similar to a diverticulum and a defect of bile duct wall continuity. Furthermore, CT showed a long common channel of the pancreaticobiliary duct, so she was diagnosed as having spontaneous CBD perforation with pancreaticobiliary maljunction. Emergency surgery was performed that revealed a necrotic diverticulum-like change on the supraduodenal part, and a 2.5 × 1 cm perforation was found on the anterolateral wall of the CBD. Peritoneal lavage was performed, and CBD perforation was resolved with a T-tube. The patient suffered refractory intra-abdominal and retroperitoneal abscess formation and bleeding from the abdominal wall, which required a long period of postoperative management. The T-tube was removed on day 136, and the patient was transferred on day 153.

A 71-year-old woman was transported to the emergency department of another hospital because of consciousness disorder. Enhanced computed tomography (CT) showed an amount of free fluid in the peritoneal cavity mainly around the right upper abdomen without free air. Ultrasonography identified cholecystolithiasis. Paracentesis revealed intra-abdominal bilious fluid with high levels of total bilirubin (21.6 mg/dL) and amylase (8697 U/L) on biochemical examination. She was diagnosed as having severe peritonitis with septic shock and was transferred to our hospital for emergency surgery and intensive care management.

Case presentation

Spontaneous CBD perforation is one of the rare presentations of acute abdomen in infants and children and is extremely rare in adults. It was first described by Freeland in 1882 [13]. Either weakness in the wall of the bile duct or an increase in intraductal pressure or both have been suggested as the cause of the perforation [11]. Fragility of the bile duct wall is considered to be caused by choledochal cysts [14, 15], pancreatitis [15, 16], chronic infection [12], congenital weakness [11], ischemia [11], pancreatic reflux [11], and torsion of the gallbladder [11]. In contrast, reported causes of perforation with increased intraductal pressure include biliary sludge or stones [1,2,3,4,5,6,7,8,9,10], tumors [11], congenital stenosis of the ampulla of Vater [11], spasm of sphincter of Oddi [11], protein plugs [11], and parasites [11]. Diagnosis of the pathogenesis is difficult and delayed, because it sometimes occurs idiopathically.

Spontaneous common bile duct (CBD) perforation is an extremely rare disease in adults. We report an adult case of CBD perforation due to choledocolithiasis accompanied with pancreaticobiliary maljunction, which is, to our knowledge, the first such case report based on a search using PubMed.

Case presentation

She had no history of past abdominal operations, and other past medical history included choledocholithiasis and pancreatitis. Hematological investigations on admission revealed coagulopathy, renal dysfunction, and circulatory insufficiency (Table 1), which indicated septic disseminated intravascular coagulation. Significantly high levels of serum transaminases, bilirubin, and pancreatic enzymes suggested a condition associated with biliary tract disease. As shown in Fig. 1, enhanced CT revealed that the supraduodenal CBD was markedly dilated similar to a diverticulum (arrow), and the bile duct wall had a partial defect in continuity (arrowhead). Moreover, the common channel of the pancreaticobiliary duct was long at 9.3 mm in length and seemed to be joined outside the muscular layer of the duodenal papilla (arrow) on the coronal CT view (Fig. 2). Eventually, we diagnosed biliary panperitonitis due to the spontaneous CBD perforation accompanied with congenital biliary dilatation and pancreaticobiliary maljunction. As her general condition improved following adequate primary resuscitation, she was able to undergo an emergency laparotomy to cure her septic peritonitis.

Although we could preoperatively diagnose CBD perforation with bilious ascites and discontinuity of the bile duct wall as proven by CT in our patient, only four cases (17.4%) in this review could be diagnosed preoperatively. In these cases, the authors reported that the loss of bile duct wall continuity on CT and high bilirubin levels in ascites were important diagnostic factors [4, 16, 23, 24]. Although most of the other cases were diagnosed by exploratory laparotomy, the perforation site in some cases could not be identified intraoperatively and required subsequent re-operation [5] or endoscopic retrograde cholangiopancreatography [3] for diagnosis. Notably, in a few cases, the perforation site could not be identified intraoperatively despite bile duct perforation being suspected preoperatively [1, 3]. In our summary, the most common site of perforation is the supra-duodenum (50%), followed by the junction of the cystic duct (22.7%). It may be helpful to observe these predominant sites when the perforation cannot be detected intraoperatively.