Pleurobiliary fistula is a rare event usually presented in patients with liver hydatid disease. Hydatid cyst may rupture in the pleural space after invasion of the diaphragm. Two major factors are required such as the mechanical effect of an increased intraluminal pressure of the biliary system due to bile duct obstruction and the local inflammatory process triggering adhesion between the liver and the diaphragm. Bronchobiliary fistula was reported also in patients with large HCCs when they underwent TACE. In the present case invasion of the common bile duct from the HCC resulted in common bile duct obstruction with subsequent increase of the intrabiliary pressure. The formation of the subphrenic liver abscess was attributed to TACE. These predisposing factors led to rupture of the abscess in the pleural space. Invasion of the bile ducts due to HCC is a rare clinical picture occurred in 2% of all HCC cases. Intrabiliary tumor growth is related with necrosis and bile duct obstruction from devitalized fragments. This devastating situation correlates with early death as reported by Kojiro and associates.
A diagnosis of bronchobiliary and pleurobiliary fistula is based on radiologic imaging findings. CT scan is non-specific to identify the biliopleural communication. Cholangiography and Tc-HIDA scan are the techniques of choice to establish the diagnosis.
Treatment of broncho- and pleurobiliary fistula is surgical [4] although few patients tolerate surgical intervention, but it can also be treated with conservative measures [5]. Tumor resection along with the fistulous tract is recommended. Segmental lung resection is also advised in cases of bronchobiliary fistula as recently reported. Malnutrition and sepsis restrict the efficacy of such a demanding operation and a recent shift to non-surgical interventions has been described in the literature by endoscopic stent placement [5]. Palliative methods offered a short term improvement followed by fistula recurrence due to failure of these methods to address the mechanism of pleurobiliary fistula formation.
Death is almost inevitable in these cases because of respiratory failure or disease progression.
In our case, chest tube drainage of the pleural effusion documented the diagnosis of pleurobiliary fistula, which was further confirmed by ERC. Due to persistence of chest tube drainage, a stent was placed initially to decompress the biliary system followed by pleurodesis.
Because of the poor liver function and sepsis, the patient eventually died; although pleural drainage was decreased with the previous mentioned conservative measures.