Bilothorax: A Rare Cause of a Pleural Effusion

First Author(s)

• NC

  Nickolas Coombs, DO

  Doctor
  University of Connecticut, United States

  Disclosure information not submitted.

Co-Author(s)

• SN

  Suong Nguyen, MD, MBA

  Doctor
  University of Connecticut, United States

  Disclosure information not submitted.
• JK

  Jennifer Kanaan, MD

  Doctor
  University of Connecticut, United States

  Disclosure information not submitted.

Title: Bilothorax: A Rare Cause of a Pleural Effusion

Case Report Body:

Introduction:
Pleural effusions (PLE) are common phenomena and are often benign. Diagnostic analysis of pleural fluid can yield information about underlying infection, known as empyema, or unclear pathology1. A rare cause of empyema is biliary translocation causing bilothorax. Presented is a case of bilothorax in the setting of recent abdominal surgery.

Description:
A 75-year-old female with a medical history of pancreatic neuroendocrine tumor (NT) presented with shortness of breath. Two months prior she was evaluated for NT resection, but developed acute cholangitis and had a cholecystectomy and hepaticojejunostomy. Postoperative complications included intra-abdominal bile leak and pulmonary embolism resulting in percutaneous transhepatic catheter placement and initiation of anticoagulation; however, due to a perihepatic hematoma she was discharged on aspirin.
On admission, she presented with a massive pulmonary embolism (PE) and received half-dose of tissue plasminogen activator. In addition to PE, a computed tomography angiogram of the chest revealed a loculated right PLE leading to a right-sided thoracentesis and chest tube placement (CTP). Pleural fluid showed a lactate dehydrogenase >7,500 U/L and a total bilirubin level of 2.6 mg/dL; serum total bilirubin was 0.5 mg/dL. Fluid cultures speciated Enterobacter cloacae and Enterococcus faecalis for which vancomycin and meropenem were started. Inferior vena cava filter was placed for acute deep vein thrombosis of the lower extremities and repeat imaging revealed resolution of PLE. She was discharged on ciprofloxacin and metronidazole for a 6-week course and therapeutic lovenox.

Discussion:
Bilothorax is a rare etiology of PLE with most cases associated with manipulation of the biliary tree or liver3. Bile salts may cause a severe inflammatory response with rapid deterioration, thus diagnosis and treatment is crucial4. A pleural fluid bilirubin to serum bilirubin (PF/S) ratio greater than 1 is highly suggestive of bilothorax,4. Treatment involves CTP for infection control and broad-spectrum antibiotics; de-escalation is guided by fluid culture. In cases of biliary-pleural fistulas surgical intervention may be needed. However, there are no guidelines on treatment for bilothorax with treatment largely anecdotal through case reports warranting further investigation.