doi 10.1308/147870810X12699662980637
On-line Case Report
A dangerous pleural effusion SOMPRAKAS BASU, SHILPI BHADANI, VIJAY K SHUKLA Department of General Surgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi, India
ABSTRACT Bilothorax is a rare complication of biliary peritonitis and, if not treated promptly, can be lifethreatening. We report a case of a middle-aged woman who had undergone a bilio-enteric bypass and subsequently a biliary leak developed, which finally led to intra-abdominal biliary collection and spontaneous bilothorax. The clinical course was rapid and mimicked venous thromboembolism, myocardial infarction and pulmonary oedema, which led to a delay in diagnosis and management and finally death. We high-light the fact that bilothorax, although a rare complication of biliary surgery, should always be considered as a probable cause of massive effusion and sudden-onset respiratory and cardiovascular collapse in the postoperative period. A chest X-ray and a diagnostic pleural tap can confirm the diagnosis. Once detected, an aggressive management should be instituted to prevent organ failure and death. Keywords: Bilothorax – Bilio-enteric bypass – Pleurobiliary fistula Accepted 13 April 2010; published online 4 June 2010
After the first description that ascitic fluid may lead to pleural effusion, there have been reports showing that air, blood or ascitic fluid1 can potentially traverse the diaphragm to enter the pleural space. Entry of bile from the abdominal cavity into the pleural space is a rare condition with high morbidity and mortality and needs urgent intervention. We report a case of biliary peritonitis leading to bilothorax and finally death in a patient who had undergone bilio-enteric bypass. Case history A 45-year-old diabetic woman presented with upper abdominal pain, jaundice and loss of appetite for 2 months. An abdominal ultrasonographic scan showed a thickened and contracted gallbladder containing
gallstones and a dilated common bile duct with mildly dilated intrahepatic biliary radicals. Her liver function tests showed an obstructive jaundice picture and a contrast-enhanced computerised tomogram (CT) suggested gallbladder malignancy. Based on the above findings, she underwent an open cholecystectomy. Operative findings were Mirrizzi’s syndrome type IV with impacted high common bile duct stones, for which cholecystectomy and a hepaticojejunostomy was performed. Postoperatively, she was doing well till the fifth postoperative day when she developed a bile leak, jaundice and features of an intra-abdominal biliary collection. Repeated ultrasound-guided percutaneous aspirations were done and parenteral nutritional support was started. Despite a brief initial response, her jaundice increased and she progressively developed bilateral pedal oedema,
Correspondence to: Somprakas Basu, Assistant Professor, Department of General Surgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi 221005, India T: +91 542 6703383; F: +91 542 2368163; E:
[email protected] Ann R Coll Surg Engl 2010; 92
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Figure 1 Chest X-ray showing right-sided massive pleural effusion due to bilothorax.
minimal pleural effusions and systemic features of sepsis. On the 14th day, she developed sudden-onset chest pain, tachypnoea, tachycardia, hypotension and a fall in arterial oxygen tension and oxygen saturation. Venous thromboembolism (VTE) or myocardial infarction (MI) were suspected. She was transferred to the intensive care unit where high flow oxygen and other supportive measures were started. VTE and MI were excluded by Doppler ultrasonographic examination, D-dimer and a negative troponin-T. However a chest X-ray showed massive, rightsided, pleural effusion (Fig. 1). When a diagnostic pleural tap from the right chest revealed bile, an intercostal tube drain was immediately inserted and 2.5 l of bile was drained. She was put on ventilatory support and the drainage decreased to about 300–400 ml/day. However, her condition progressively deteriorated with the onset of respiratory distress syndrome (ARDS) and multi-organ failure and she died on the 21st postoperative day. Discussion The proposed mechanisms of a bilothorax include direct extension of a biloma through the diaphragm or through a congenital defect1 or following traumatic diaphragmatic perforations.2 It has been reported to occur after spontaneous gallbladder perforation,3 open cholecystectomy,4 percutaneous biliary drainage5 and as a sequel of biliary peritonitis following jejunal, gastric or bile duct perforations.1,6,7 The infected bile erodes through the diaphragm, most commonly through the posterior recess or through a pre-existing defect, and the condition usually presents with features of shock and sudden cardiorespiratory collapse.
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A dangerous pleural effusion
The acute onset of cardiorespiratory symptoms led us to consider VTE and MI, in the background of predisposing factors like prolonged bed-ridden state, diabetes, and sepsis. The onset of pedal oedema at 2 weeks was also suggestive of a probable deep venous thrombosis. Sixteen hours elapsed before bilothorax was diagnosed. Awareness of this possibility is an important prerequisite to its early diagnosis. It is usual to consider VTE, MI or pulmonary oedema as the probable causes of sudden-onset respiratory distress with chest pain in a postoperative patient. However, when similar events are encountered in a patient following biliary surgery or biliary peritonitis, we stress that the clinician should also consider a bilothorax, which manifests clinically as a massive pleural effusion. A portable chest X-ray is the simplest investigation to confirm pleural effusion. An early diagnostic tap of a rapidly developing pleural effusion will confirm the diagnosis of biliary pleural effusion. Aggressive management is justified, as the thick bilious fluid urgently needs an intercostal tube drain. Moreover, once diagnosed, the patient should be considered for early positive pressure ventilation as infected bile in the pleural space can rapidly lead to ARDS. Consideration should be given to early abdominal intervention to deal with the persisting bile leak, which may prevent progressive deterioration. Conclusions Bilothorax as a complication of biliary peritonitis is rare and is difficult to diagnose as it may mimic other postoperative cardiorespiratory complications. Awareness is important for early diagnosis of this ominous condition. A chest X-ray and a diagnostic pleural tap are all that is necessary to confirm the diagnosis, although other cardiorespiratory catastrophes should always be excluded. An early and aggressive management is required to prevent organ failure and loss of life. References 1. Rowe PH. Bilothorax – an unusual problem. J R Soc Med 1989; 82: 687–8. 2. Ivatury RR, O’Shea, Rehman M. Post traumatic thoracobiliary fistula. J Trauma 1984; 24: 438–42. 3. Cunningham LW, Grobman M, Paz ML, Hanlore CA, Promisloff RA. Cholecystopleural fistula with cholelithiasis presenting as a right pleural effusion. Chest 1990; 97: 751–2. 4. Prabhu R, Bavara C, Purandare H, Supe A. Pleurobiliary fistula – a delayed complication following open cholecystectomy. Indian J Gastroenterol 2005; 24: 28–9. 5. Strange E, Allen ML, Freedland PN, Cunningham J, Sahn SA. Pleural fistula as a complication of percutaneous biliary drainage: experimental evidence for pleural inflammation. Am Rev Respir Dis 1988; 137: 959–61. 6. Bini A, Grazia M, Petrella F, Stella F, Bazzocchi R. Spontaneous biliopneumothorax (thoracobilia) following gastropleural fistula due to stomach perforation by nasogastric tube. Ann Thorac Surg 2004; 78: 339–41. 7. Ramnarin IR, Murpur AK, McMohan MJ, Thorpe JAC. Pleurobiliary fistula from ruptured choledochal cyst. Eur J Cardiol Thorac Surg 2001; 19: 216–8.
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