Golden sputum: a case of acquired bronchobiliary fistula

SESSION TITLE: Lung Pathology: Atypical Connections SESSION TYPE: Rapid Fire Case Reports PRESENTED ON: 10/09/2023 02:10 pm - 02:55 pm INTRODUCTION: Bronchobiliary fistulas (BBF), an anomalous track between the biliary and the bronchial tree were first described as a complication of hydatid cyst in 1850. Since then, BBF has been described in congenital defects, biliary tract obstruction, complications after traumatic injury, and surgery. BBF can present with bilioptysis (presence of bile in the sputum), an infrequent cause of chronic cough yet highly suggestive of BBF. We present a case of BBF in a patient with protracted treatment for recurrent hepatocellular carcinoma. CASE PRESENTATION: A 77-year old woman presented to the emergency department with green-colored productive cough for four weeks. She had a history of Stage IV hepatocellular carcinoma (HCC) treated first with radiofrequency ablation with recurrence requiring hepatic lobectomy. Computed tomography scan of the abdomen showed a right hepatic biloma site probably communicating with right pleural space (Figure 1). Subsequent investigation with hepatobiliary iminodiacetic acid (HIDA) scan was done and revealed radiotracer uptake in the right mainstem bronchus, trachea, and left mainstem bronchus (Figure 2). She was found to have a biloma with concerns for fistulization to the right main bronchus. Interventional radiology placed a right hepatic lobe 8 french (F) drain at the prior surgical site. Bronchoscopic examination was noted to have copious amounts of yellow-tinged sputum consistent with bilious content. An abscessogram with interrogation of biloma and bronchogram confirmed a bile leak at the right hepatic lobectomy site with diaphragmatic and pulmonary erosion and with the creation de-novo of a broncho-biliary fistula (BBF). Subsequently, the patient underwent endoscopic retrograde cholangiopancreatography (ERCP) with papillotomy biliary stent placement and drainage. Contrast instilled into the common bile duct (CBD) revealed its communication to the biloma via the biliary tree intrahepatic branches. Further respiratory deterioration required mechanical ventilation and patient ultimately was transitioned to hospice and expired. DISCUSSION: Bronchobiliary fistula (BBF) is an out of the ordinary existing condition with bilioptysis being the hallmark sign. Our patient's BBF originated as a complication from hepatocellular carcinoma treated with radiofrequency and Nano-knife ablation, hepatic lobectomy and bleeding requiring arterial embolization. The habitual clinical presentation of BBF is bilioptysis or bile-stained sputum. Bile backflow can precipitate serious chronic irritable cough, respiratory failure such as acute bronchiolitis, chronic bronchopneumonia, bronchiectasis, mediastinitis and severe necrotizing pneumonia. Therefore, immediate diagnosis and treatment is critical for prognostication. If the diagnosis remains ambiguous, the appearance of bilirubin in the sputum sample can indicate BBF as crystallization of bilirubin in bronchoalveolar lavage as been described as a worthwhile marker in previous case reports. CONCLUSIONS: Management of BBF poses a clinical challenge and is associated with high morbidity and mortality. Less invasive methods are preferred, but there are no clear-cut approach when treating this entity. An interdisciplinary approach is essential for the prompt diagnosis and treatment of a BBF and can aid in proper prognostication. REFERENCE #1: Peacock TB. Case in Which Hydatids Were Expectorated, and One of Suppuration in a Hydatid Cyst of the Liver Communicating with the Lungs. Edinb Med Surg J. 1850;74(184):33-46. https://www.ncbi.nlm.nih.gov/pubmed/30331080 REFERENCE #2: Hai S, Iimuro Y, Hirano T, Suzumura K, Yada A, Fujimoto J. Bronchobiliary fistula caused after hepatectomy for hepatocellular carcinoma: a case report. Surg Case Rep. 2016;2(1):147. doi:10.1186/s40792-016-0273-z REFERENCE #3: Lazarou V, Moris D, Papalampros A, Tsilimigras DI, Karachaliou GS, Petrou A. Bronchobiliary fistula after hepatectomy: A case report and review of the literature. Mol Clin Oncol. 2019;11(6):602-606. doi:10.3892/mco.2019.1935 DISCLOSURES: No relevant relationships by Jose Acosta-Rullan No relevant relationships by Christian Almanzar Zorrilla No relevant relationships by Mauricio Danckers No relevant relationships by Aunie Danyalian No relevant relationships by Raiko Diaz No relevant relationships by Daniel Heller No relevant relationships by Danay Herrera No relevant relationships by Manjot Malhi No relevant relationships by Rafael Miret