Eur J Cardiothorac Surg 2001;19:216-218
© 2001 Elsevier Science NL
Pleuro-biliary fistula from a ruptured choledochal cyst
I.R. Ramnarinea,
A.K. Mulpura,
M.J. McMahonb,
J.A.C. Thorpea
a Department of Thoracic surgery, Yorkshire Heart Centre, Leeds General Infirmary, Great George Street, Leeds, LS1 3EX, UK
b Leeds Institute for Minimally Invasive Surgery, Leeds General Infirmary, Great George Street, Leeds, LS1 3EX, UK
Received 6 June 2000;
received in revised form 31 October 2000;
accepted 15 November 2000.
Corresponding author. Tel.: +44-113-392-5897; fax: +44-113-392-8092
e-mail: ianrramnarine{at}hotmail.com
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Abstract
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We present a case of rupture of an intrahepatic choledochal cyst through the diaphragm resulting in a pleuro-biliary fistula and a right pleural empyema which was surgically treated. Hepatobiliary complications resulting in biliary empyema of the pleura are discussed.
Key Words: Choledochal cyst rupture • Pleuro-biliary fistula • Empyema
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1. Case presentation
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A 78-year-old female presented with a 6-month history of right upper quadrant pain and nausea. On investigation with abdominal ultrasound and computer tomography the patient was found to have a large (10x9x7 cm) hepatic cyst which was compressing hepatic tissue, dilated biliary ducts and a shrunken gallbladder. Endoscopic retrograde cholangiopancreatography (ERCP) demonstrated a grossly enlarged common bile duct with no obvious cause; a sphincterotomy was performed. The patient continued to feel unwell and then had a laparoscopic cholecystectomy and fenestration of the hepatic cyst with intra-operative cholangiogram and ultrasound. Histological examination demonstrated a choledochal cyst wall, hepatic cirrhosis and no malignant cells. The patient went home and her condition improved. Even though the choledochal cyst was still seen on sequential imaging (Fig. 1), she was unwilling to undergo further surgery.
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Fig. 1. Magnetic resonance cholangiopancreatogram of a coronal section through the liver. Bile appears white. The dilated biliary tree is shown with a large intrahepatic choledochal cyst pointing at the dome of the diaphragm. The cyst is compressing hepatic tissue. (Key: CC, Choledochal Cyst; D, Diaphragm).
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Two years after the initial operation the patient was admitted with fever, dyspnoea and pleuritic right chest pain. She was febrile and tachypnoeic. Chest examination revealed decreased right-sided expansion, dullness on percussion, decreased air entry and coarse crepitations of the right base and associated abdominal tenderness in the right upper quadrant. Chest X-ray showed right middle and lower lobe consolidation and a moderate sized right pleural effusion. Blood tests revealed normal urea, electrolytes, haemoglobin and platelet count, but the neutrophil count, serum bilirubin and alkaline phosphatase were raised. A right-sided intercostal drain was inserted and initially drained 1500 ml of bile-stained fluid. The patient was started on antibiotics. Examination of the pleural fluid demonstrated a high bile content, but no organisms could be seen or cultured. The drainage persisted and the patient was referred for surgery.
A right thoracotomy revealed a cortex of thickened pleura surrounding a cavity full of bile, resembling an empyema. Bile was seen to be coming from an opening in the right diaphragmatic dome (Fig. 2). The area under the diaphragm was explored and a fistula connecting the pleura and the intrahepatic cyst was found. The cortex was removed and the lung mobilised; the cyst was evacuated. Talc was insufflated into the pleura and cyst and both were drained. Non-absorbable sutures were used to close the fistula. Following the operation there was clinical improvement and lung re-expansion, but bile continued to drain from the cyst. An abdominal ultrasound demonstrated dilated intra- and extra-hepatic biliary ducts. Endoscopic retrograde cholangiopancreatography was performed with stenting of the common bile duct. Gradually the cyst shrank and disappeared. Because of the effects of chronic illness and prolonged malnutrition the patient was slow to recover and mobilise, but went home without complications.
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Fig. 2. Colour photograph showing right thoracotomy with lung retracted. Bile can be seen flowing from beneath the diaphragm and through the pleuro-biliary fistula. (Key: D, diaphragm; F, fistula opening; B, bile).
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2. Discussion
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A choledochal cyst is an enlargement of the biliary tree, in either an intra- or extra-hepatic location. It is more common in females (4:1), and may be complicated by cholelithiasis and cirrhosis. Type I is the most common form, characterised by fusiform dilatation of the common hepatic and common bile duct with the cystic duct entering the choledochal cyst. Abdominal ultrasound is sufficient in most cases for the diagnosis. Cystoduodenostomy and cystojejunostomy are no longer recommended as treatment options. The operation of choice is complete resection of the cyst and a Roux-en-Y hepatojejunostomy. Carcinoma, cholangitis, anastomotic stricture and progressive liver disease may complicate surgery [1].
Choledochal cyst rupture is uncommon, but rupture into the abdomen may occur following trauma. A single case of choledochal cyst rupture into the retroperitoneum has also been reported [2]. A review of the literature reveals that choledochal cyst rupture through the diaphragm has not been previously described, making our case unique.
This case presented as an empyema and was treated with decortication and drainage. A thoracoscopic approach would not have allowed us to explore the subdiaphragmatic pathology or to perform decortication safely, but is a reasonable alternative to a full thoracotomy.
Bile found in the chest is a rare occurrence. It most commonly complicates surgery for oesophageal disease, especially when anastomoses leak. Other major causes include thoraco-biliary fistulas from trauma [3] and hepatic hydatid cyst rupture through the diaphragm [4]. When a patient presents with bile in the chest and there is a previous history of biliary surgery, the development of surgical complications must be suspected, examples include subdiaphragmatic abscesses [5] or biliary stones that were not removed at surgery and that have migrated [6,7]. Conservative management of a thoraco-biliary fistula is unlikely to be successful because bile acts as an irritant and impairs wound healing, especially if bile flow continues. Surgical closure of the fistula with adequate drainage of bile is the option most likely to work.
The message from this case is to beware of bile in the chest and judicious surgical intervention is the preferred option.
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References
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Abstract
1. Case presentation
