Case Presentation: A 51-year-old female with a 15-year history of alcohol use disorder (approximately one pint of vodka daily) presented with nausea, vomiting, abdominal pain, and jaundice. On arrival to the emergency department, she was febrile and hypoxic. Initial workup revealed evidence of cirrhosis with esophageal varices (without ascites) and bibasilar airspace opacities. She was admitted for management of decompensated alcoholic cirrhosis and severe sepsis secondary to community-acquired pneumonia. Despite treatment with broad-spectrum antibiotics (piperacillin-tazobactam and doxycycline), her respiratory and hemodynamic status deteriorated, necessitating intubation, mechanical ventilation, and vasopressor support. By the third week of admission, due to persistent respiratory failure, antibiotics were escalated to meropenem and doxycycline. Bronchoscopy revealed diffuse yellowish secretions concerning for bilioptysis, although no mucus plugs or purulent secretions were observed. Sputum bile levels were positive, measuring 0.5 mg/dL.Following gradual improvement in respiratory status, the patient was extubated and transferred out of the ICU. However, she required re-intubation after developing ARDS in the setting of recurrent sepsis. Repeat bronchoscopy again showed diffuse yellowish secretions however with new purulent sputum that grew MRSA on cultures. Despite multiple courses of broad-spectrum antibiotics, diuretics, and steroids, she could not be weaned off mechanical ventilation. Notably, serum hyperbilirubinemia persisted throughout her hospitalization, peaking at 14.2 mg/dL. After a prolonged two-month hospital course, including tracheostomy and gastrostomy tube placement, she was discharged to a long-term acute care facility.
Discussion: Presence of bile in the sputum, or bilioptysis, is a rare clinical finding most commonly associated with bronchobiliary fistula, typically secondary to trauma, hepatic abscess, carcinoma, or procedural complications related to hepatocellular carcinoma (HCC).1,2 Fewer than 50 cases have been reported in the literature, the majority involving anatomic communication between the biliary and bronchial systems.1 In our patient, bronchoscopy revealed diffuse golden-yellow secretions without any evidence for bronchobiliary fistula, raising suspicion for an alternate etiology. Severe ARDS and sepsis, in the context of underlying alcoholic hepatitis and cirrhosis, likely led to disruption of the alveolar-capillary barrier and therefore diffusion of bilirubin into the alveoli. This mechanism is supported by prior reports of bilioptysis in patients with ARDS and sepsis in the absence of fistula formation.3,4 Decompensated cirrhosis itself promotes systemic inflammation and increases pulmonary capillary permeability, facilitating diffusion of bilirubin along a concentration gradient into alveolar spaces.5 Additionally, septic endotoxemia has been shown to disrupt the pulmonary endothelial glycocalyx—a key barrier regulating vascular permeability—further supporting increased capillary leak as a plausible mechanism.6,7
Conclusions: Bilioptysis is an exceptionally rare clinical finding, with fewer than 50 cases having been reported.1,2 Non-fistulous causes of bilioptysis are far less frequently described, observed in the setting of severe systemic illness.3, 4, 5 This report presents a case of bilioptysis occurring in the context of critical illness and decompensated liver disease.