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Congenital bronchobiliary fistula in a 65-year-old woman
Hideshi Uramoto1, Osamu Kawano, Mineharu Sugimoto
1Kumamoto Saishunso National Hospital, National Hospital Organization, Kosi.
Insights
Congenital bronchobiliary fistula (CBBF), a rare condition, was incidentally found in a 65-year-old woman. This case represents the oldest reported adult diagnosis of CBBF, likely due to a narrow fistula.
Area of Science:
- Medical Diagnostics
- Congenital Malformations
- Pulmonology
- Hepatology
Background:
- Congenital bronchobiliary fistula (CBBF) is a rare congenital anomaly, typically diagnosed in infancy due to respiratory issues.
- Surgical intervention is the standard treatment for CBBF.
- Most reported cases occur in neonates and infants, with few adult diagnoses.
Observation:
- A 65-year-old woman presented with incidental findings of CBBF during investigations for pulmonary nodules.
- Bronchoscopy revealed an accessory bronchus with bilirubin-rich secretions.
- Thoracic CT, drip infusion cholangiography, and cholescintigraphy confirmed the airway-biliary tract communication.
Findings:
- The patient had a narrow congenital bronchobiliary fistula.
- The narrowness of the fistula is hypothesized to be the reason for the patient's asymptomatic long survival.
- This case is the fifth reported adult diagnosis and the oldest at 65 years.
Implications:
- This case expands the known age range for adult diagnosis of CBBF.
- It highlights the possibility of late diagnosis in adults with CBBF, particularly if the fistula is narrow.
- Further research into the long-term implications and management of incidentally discovered CBBF in adults is warranted.
Abstract:
Congenital bronchobiliary fistula (CBBF) is quite a rare malformation and the diagnosis is usually made within a few hours or years from birth because of lower respiratory diseases beginning from early infancy. Surgical repair is necessary. Of the 29 cases reported, 4 occurred in adults aged 22-32 years. We detected CBBF incidentally in a 65-year-old woman. During bronchoscopy and thoracic computed tomographic study of the pulmonary nodules, we found an accessory bronchus descending from the carina and composed of a dark green secretion that contained 10% bilirubin. Drip infusion cholangiography revealed air in the left bile duct. Cholescintigraphy showed dilatation of the left bile duct and radiotracer pooling at the top edge of the left hepatic lobe. These findings indicated a narrow fistula between the airway and biliary duct. We attributed the patient's long survival without major complications to the narrowness of the communication. To our best knowledge, this is the fifth and oldest reported adult diagnosed with CBBF.
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