Thoracobilia: a rare complication of gallstone disease

Abstract

Thoracobilia and pleurobiliary fistulae secondary to obstructed choledocholithiasis are rare complications of gallstone disease. We present the case of a 64-year-old man with gallstone pancreatitis who developed a pleurobiliary fistula secondary to an obstructing ductal calculus, and was successfully managed non-operatively.

Thoracobiliary fistulae (TBF) are an uncommon clinical entity. Biliary contamination in the thorax (thoracobilia) can be limited to the pleural space (pleurobiliary fistula [PBF]) or can involve the pulmonary parenchyma and bronchial tree (bronchobiliary fistula [BBF]).^1,2 BBF present with bilioptysis (expectoration of bile stained fluid), which is pathognomonic.^3,4 PBF commonly presents with fever, right pleuritic chest pain, right upper quadrant and shoulder tip pain, and a persistent or delayed onset right pleural effusion.^5,6

We report an unexpected case of PBF secondary to choledocholithiasis that was successfully treated endoscopically.

Case history

A 64-year-old man, presented as an emergency with a 1-week history of feeling generally unwell and pain consistent with biliary colic. Before admission, the pain had become more severe and associated with bilious vomiting, fever, rigors and dark urine. The patient reported similar episodes of pain over the last 2 years but none required hospital admission.

On initial examination, he was jaundiced and dehydrated, but afebrile. He was tender in the right upper quadrant and epigastrium but with no peritonism. The rest of his examination was unremarkable. Initial blood tests revealed a hyperamylasaemia and obstructive jaundice.

He was given supportive care including oxygen, intravenous fluid resuscitation and his fluid balance was monitored accurately.

Abdominal ultrasonography showed a normal gallbladder with no calculi; however, the common bile duct (CBD) was dilated at 13 mm with intrahepatic duct dilatation. No intraductal calculi were seen.

The following day (48 h post-presentation), the patient became significantly hypoxic. Clinical examination revealed decreased air entry with dullness to percussion on the right side of the chest. A chest radiograph showed a new large right pleural effusion (Fig. 1). A Seldinger chest tube was inserted under ultrasound guidance, and immediately drained a 1 l of bile and continued to drain approximately 100–200 ml/day. The diagnosis of a TBF was confirmed by biochemical demonstration of elevated bilirubin level of 180 μmol/l in the pleural aspirate.

Figure 1.

Chest radiograph showing a new large right pleural effusion 48 h post presentation.

MRCP was then performed and demonstrated sludge in the fundus of the gallbladder with a dilated CBD of 14 mm with a filling defect in the distal CBD and distal end of the pancreatic duct; the pancreas looked normal. The diagnosis of a PBF was made with a definite fistulous tract identified between the left lobe of the liver and the right hemidiaphragm (Fig. 2A–C).

Figure 2.

(A) MRCP cholangiogram showing the right pleurobiliary fistula. (B) MRCP cholangiogram showing a dilated CBD and a gallstone distally. (C) MRCP cholangiogram showing a calculus in the pancreatic duct.

An ERCP was performed. The papilla was stenosed, scarred and not successfully cannulated, but the CBD could be seen to bulge into the duodenum above the papilla. An extended needle-knife sphincterotomy was carried out at this site and a cholangiogram demonstrated a single obstructing stone within the CBD. Occlusion cholangiogram demonstrated a small fistula from the biliary tree into the pleural space (Fig. 3). The CBD was dredged and the stone was seen to emerge. The patient made good progress post-procedure with the chest tube in situ, inflammatory markers falling and liver function tests normalising.

Figure 3.

A cholangiogram demonstrating the small pleurobiliary fistula with contrast collecting around the tube thoracostomy.

The chest tube remained in situ for 10 days until minimal output was measured and the patient was then discharged home. Follow-up has been arranged in order to discuss the need for an elective laparoscopic cholecystectomy.

Discussion

Thoracobilia has been previously reported as a complication of choledocholithiasis,^1,7 although the commonest cause in Western society is due to trauma and missed hepatobiliary injury.^3,7

A high index of suspicion, prompt diagnosis and treatment is crucial for the management of PBF. Bile has a corrosive effect upon the lung and pleural space¹ and persistent flow can cause severe lung damage (e.g. pleural fibrosis, pneumonitis, pulmonary fibrosis).²

Diagnosis is made primarily on clinical history and symptoms. Treatment is based on the standard management of any fistula, in that all fistulas will eventually close if there is no associated infection, malignancy or distal obstruction. Although the optimal treatment of PBF is still debated, a minimally invasive approach is preferred because of the high morbidity associated with thoraco-abdominal surgery.

MRCP remains the investigation of choice to identify the cause and site of any biliary obstruction and delineate the fistulous communication.⁷ ERCP and percutaneous transhepatic cholangiography (PTC) are useful for diagnostic and therapeutic purposes in cases of PBF due to cholelithiasis.^1,3 ERCP has the obvious benefits of being able to apply therapeutic procedures such as stent insertion, dilatation of the biliary ducts, removal of gallstones and sphincterotomy.^4,5

Initial treatment is conservative in the form of intercostal drainage, drainage of any other collections, rehydration, correction of any metabolic abnormalities,⁷ subcutaneous administration of octreotide, enteral nutrition and broad-spectrum antibiotics.² Biliary decompression is mandatory to facilitate closure of the fistulous tract and this is done best by ERCP.⁷ Singh et al.² recommend endoscopic sphincterotomy when there is persistence of symptoms within 72–96 h of the placement of tube thoracostomy or percutaneous drainage of sepsis.

Conclusions

No guidelines exist for the treatment of pleurobiliary fistula; therefore, management should be tailored to individual patient needs. Endoscopic therapies to relieve biliary obstruction and achieve drainage can obviate the need for high-risk surgery.