Introduction:
Bronchobiliary fistulas (BBFs) are abnormal connections between the bronchial system and the biliary tree. These fistulas, though rare, pose significant clinical challenges. BBFs can be caused by various factors including liver infections, congenital conditions, abscesses, malignancy, and trauma. The hallmark symptom is bilioptysis, a cough producing bile, which can lead to severe bronchial irritation and bronchiectasis. This study focuses on uncommon and delayed presentations of BBFs following liver-related interventions including surgical resection, radioembolization, and radiofrequency ablation, diverging from the typical immediate or short-term development post-procedure, thereby aiming to enhance clinicians' understanding for improved monitoring and management.

Methods:
A retrospective review was performed using a data access web application to identify electronic medical records from January 2000 to October 2023 using ICD-10 codes. Inclusion criteria included patients with a history of liver diseases or procedures who developed delayed onset BBFs, defined as the development of fistula symptoms or diagnosis occurring more than six months post-intervention. Exclusion criteria included patients with BBF onset within 6 months post intervention. 330 cases of BBF were identified, and 5 met our inclusion criteria for delayed BBF. Charts were abstracted to identify the time interval between liver-related procedures and the onset of BBF, the symptoms, demographic information, treatment, and outcomes.

Results:
The patients ranged from 57 to 78 years of age, with diverse backgrounds and medical histories. The onset of BBF symptoms varied significantly, with intervals ranging from a 1 year to 5 years post intervention, occurring well beyond the typical post-procedure period. 3/5 patients had underlying hepatobiliary cancer and presented with biliptysis, 4/5 patients had normal bilirubin, 3/5 patients had a CT scan, 4/5 patients had bronchoscopy and 2/5 received MRI. Treatment strategies primarily involved endoscopic methods, with all patients receiving ERCP accompanied by stenting as the principal approach. This method proved successful in alleviating symptoms in 4/5 of the cases studied. One patient did exhibit a more severe progression of the disease, necessitating the implementation of multiple invasive surgical interventions. This included lobectomy and pneumectomy. Unfortunately, despite these measures, the patient expired due to their complications.

Conclusion:
This study highlights the diverse range of presentation of BBFs in patients with a history of liver disease or surgery, and details that ERCP with stenting can often successfully treat these complications.

Details of the cases

Mohamad Elgozair

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