THE AMERICAN JOURNAL OF GASTROENTEROLOGY Copyright © 1998 by Am. Coll. of Gastroenterology Published by Elsevier Science Inc.
Vol. 93, No. 11, 1998 ISSN 0002-9270/98/$19.00 PII S0002-9270(98)00517-6
Spontaneous Rupture of an Intrahepatic Bile Duct With Biloma Treated by Percutaneous Drainage and Endoscopic Sphincterotomy Hidetoshi Fujiwara, M.D., Masahiro Yamamoto, M.D., Masanori Takahashi, M.D., Hidefumi Ishida, M.D., Osamu Ohashi, M.D., Hirohiko Onoyama, M.D., Yoshifumi Takeyama, M.D., and Yoshikazu Kuroda, M.D. First Department of Surgery, Kobe University, School of Medicine, Kobe, Japan
A case of spontaneous rupture of an intrahepatic bile duct with biloma formation treated by percutaneous drainage and endoscopic sphincterotomy is reported. A 73-yr-old woman was admitted with fever and abdominal pain. There was no past history of abdominal surgery, instrumentation, or trauma. Ultrasound and computed tomography revealed a massive fluid collection in the abdominal cavity. Endoscopic retrograde cholangiography demonstrated extravasation of contrast medium from a distal biliary radicle in the left lobe of the liver. After successful treatment by percutaneous drainage and endoscopic sphincterotomy, the patient did well. Ultrasound and computed tomography showed resolution of the biloma. Nontraumatic bilomas are very rare: in fact, only 24 cases of spontaneous biloma have been reported. Endoscopic treatment for patients with spontaneous bilomas can be safe and effective, and should be considered. (Am J Gastroenterol 1998;93:2282–2284. © 1998 by Am. Coll. of Gastroenterology)
aminations revealed a white blood cell count of 12,500/ mm3, a total bilirubin concentration of 1.8 mg/dl (normal ,1.0 mg/dl), an alkaline phosphatase activity of 501 IU/L (normal 100 –303 IU/L), and a gamma glutamyl transpeptidase activity of 206 IU/L (normal 9 – 64 IU/L), a blood urea nitrogen concentration of 37 mg/dl (normal 9 –22 mg/ dl), and a C reactive protein concentration of 22.99 mg/dl (normal ,0.3 mg/dl). The electrolytes, total protein concentrations, and serum amylase activity were normal. Ultrasonography (US) revealed dilated extrahepatic and intrahepatic bile ducts and a massive fluid collection on the left side of the abdomen. Multiple septations were noted (Fig. 1). Computed tomography (CT) revealed stones in both the extrahepatic and intrahepatic bile ducts (Fig. 2). In addition, a fluid collection was noted in the left upper abdomen. A percutaneous catheter was inserted into the collection under ultrasound guidance, through which 200 ml of dark brown material was drained. Cultures of the drainage revealed mixed growth of coliform bacteria. Laboratory analysis of the drainage demonstrated a total bilirubin concentration of 22.9 mg/dl, an alkaline phosphatase activity of 140 IU/L, and an amylase activity of 49 IU/L. One day after drainage the volume of drainage decreased to 20 ml/day. Endoscopic retrograde cholangiopancreatography (ERCP) demonstrated stones in the common bile duct (CBD) and extravasation of contrast medium from a distal biliary radicle in the left lobe of the liver (Fig. 3). After endoscopic sphincterotomy, the stones in the CBD were extracted with a basket catheter. Over the next 3 wk, the patient did well clinically, and US and CT showed progressive resolution of the biloma. The percutaneus drainage catheter was clamped and removed on postdrainage day 22. After the percutaneous catheter was removed, the patient recovered uneventfully. She was continued on management of hypertension and discharged 6 wk after admission.
INTRODUCTION Nontraumatic rupture of the biliary tree is an uncommon event occurring mainly in the extrahepatic tract, usually in the common bile duct in the presence of biliary stones (1). The occurrence of a nontraumatic rupture of the biliary tree involving the intrahepatic ducts and resulting in an encapsulated bile collection is very unusual (2). We report a case of nontraumatic intrahepatic rupture of the biliary tree with biloma formation treated by percutaneus drainage and endoscopic sphincterotomy. CASE REPORT A 73-yr-old woman was admitted with 4 days of fever, abdominal pain, and inability to cope at home. There was no significant past medical history except for hypertension, nor any history of abdominal surgery, instrumentation, or trauma. Her body temperature was 37.7°C. Laboratory ex-
DISCUSSION The term “biloma” was coined by Gould and Patel in 1979 to describe an encapsulated bile collection located outside the biliary tree (3). In most cases, bilomas occur
Received Oct. 3, 1997; accepted May 11, 1998. 2282
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FIG. 1. Ultrasonography of the fluid collection in the left upper abdomen demonstrating multiple loculations.
INTRAHEPATIC BILE DUCT RUPTURE
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FIG. 3. Endoscopic cholangiogram demonstrating extravasation of contrast from a distal biliary radicle in the left lobe (arrow), and stones (arrowhead) in the dilated common bile duct. TABLE 1 Characteristics of the Patients With a Spontaneous Biloma Reported Between 1979 and 1997
FIG. 2. Computed tomography revealing a fluid collection in the inferior part of the left hepatic lobe (arrowhead), and a stone in the dilated common bile duct (arrow).
secondary to traumatic or iatrogenic injury, including abdominal surgery, laparoscopic surgery, and percutaneous catheter drainage. Spontaneous perforation of the biliary tree without an antecedent history of trauma or previous surgery is an exceedingly rare cause of biloma (2). Nontraumatic bilomas have previously been reported as spontaneous bilomas. All 25 cases of spontaneous bilomas reported since 1979 have been reviewed (Tables 1 and 2) (2, 4 –15). There were 12 men and 13 women, with a mean age of 61.7 6 18.3 yr (range, 24 – 86 yr). Spontaneous bilomas were most often related to choledocholithiasis, cancer of the biliary tree, acute cholecystitis, drug administration, sickle cell disease, and tuberculosis. The site of biliary leakage was the left hepatic duct (n 5 8), the gallbladder (n 5 5), the right hepatic duct (n 5 2), or the common bile duct (n 5 1). The location of the biloma was around the left lobe of the liver (n 5 11), around the right lobe of the liver (n 5 10), or in the upper abdomen (n 5 4). Spontaneous perforation of the biliary tree is unusual, and has been linked to several potential mechanisms including increased intraductal pressure caused by spasm of the
Average Age (yr) 61.7 6 18.3 range, (24–86) Gender Male Female Underlying conditions Choledocholithiasis Cancer of the biliary tree Acute cholecystitis Hepatic infarction Hepatic abscess Nephrotic syndrome Obstructive jaundice Sickle cell disease Tuberculosis Location of the biloma Around the left lobe of the liver Around the right lobe of the liver Upper abdomen Site of biliary leakage Left hepatic bile duct Gallbladder Right hepatic bile duct Common bile duct Unknown
Age (yr)
Number of Patients
,49 50–69 .70
6 (25%) 6 (25%) 12 (50%)
12 13
(48%) (52%)
16 2 1 1 1 1 1 1 1 11 10 4 8 5 2 1 9
sphincter of Oddi or obstruction by tumor or stone, necrosis of a portion of the bile duct wall secondary to a calculus, intramural infection, rupture of a diverticulum or a cyst of the biliary tree, and focal liver infarction (2, 4, 16). Bilomas are readily detected by US or CT. Epithelization of the biloma in our case was detected on US as septations. This was not seen on CT (17). Abdominal CT is the good method to identify and localize a bile collection, and US is suitable for following the progress of a biloma once it has
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TABLE 2 Management of the Spontaneous Bilomas Percutaneous drainage Cholecystectomy and choledocholithotomy Cholecystectomy Choledocholithotomy PTCD EST Surgical drainage
15 10 2 2 1 1 1
PTCD 5 percutaneous transhepatic cholangiodrainage; EST 5 endoscopic sphincterotomy.
been identified. ERCP has been incriminated in the occurrence of biliary tract rupture (18). In this case, ERCP did not cause the rupture, as since the biloma was detected previously by US and CT. However, ERCP clearly demonstrated the presence and site of the biliary leakage. Magnetic resonance imaging (MRI) is also useful in the differential diagnosis of bilomas (19). Bilomas are heterogenously intense on T1-weighted images, and homogenously hyperintense on T2-weighted images. In management of the biloma, percutaneous catheter drainage was performed in 15 patients (Table 2). Precise localization of the biloma allows the possibility of percutaneous drainage, which can obviate the need for surgery. Epithelization of the biloma may increase the success of percutaneous aspiration of the biloma (8), although septations may prevent complete drainage. This procedure was sufficient in our case. Surgical treatment was employed in 15 patients; cholecystectomy and choledocholithotomy in 10, cholecystectomy in 2, choledocholithotomy in 2, and drainage in 2. Only our patient was treated by endoscopic sphincterotomy (Table 2). The traditional therapeutic approach has been surgical, but a number of authors have reported successful endoscopic management of bilomas, using the techniques of sphincterotomy and endoprosthesis placement (20). Traumatic bile leaks, which are most often found in postoperative patients, have also been treated by stent placement (21). Endoscopic treatment includes endoscopic sphincterotomy to lower the pressure in the biliary tree, and stone extraction. The prosthesis provides a conduit past the site of leakage and bridges any region of obstruction distal to the site of extravasation. The latter procedure was not used in our case because of the intrahepatic location of the leak and a noticeable improvement in biliary passage after extraction of the stones in the CBD. Endoscopic treatment for patients with spontaneous bilomas can be safe and effective, and should be considered.
Reprint requests and correspondence: Hidetoshi Fujiwara, M.D., First Department of Surgery, Kobe University, School of Medicine, 7-5-2, Kusunokicyo Chuoku, Kobe 650-0017, Japan.
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