We describe a case of bronchoesophageal fistula secondary to boncholithiasis not detected by swallow study prior to broncholith removal.
This 43 year old advertising consultant, with a history of hypertension and tobacco addiction, presented with one year of intermittent cough. The patient had approximately eight ounces of greenish sputum production per day with paroxysms of coughing and rare hemoptysis worst upon awakening and upon drinking. His social history was remarkable for living in Minnesota and working with manure during past summer jobs. Initial work-up included spirometry and chest radiography were normal. Empiric therapeutic interventions including successful smoking cessation, a course of antibiotics, bronchodilators, corticosteroids, and acid suppression did not alter his symptoms. A high resolution CT of the chest demonstrated small calcified mediastinal lymph nodes suggestive of remote granulomatous disease. The patients symptoms persisted and a barium esophagram showed a focal outpouching of the left lateral mid-esophageal wall with a tethered appearance adjacent to a one centimeter calcified lymph node without evidence of barium extravasation. Rigid bronchoscopy confirmed the presence of bronchioliths, and three were removed from the left main bronchus (Figure). The patient developed worsened cough after drinking. Repeat barium esophagram showed barium extending from the apex of the previously-seen diverticula into the left lower lobe bronchus. The patient underwent thoracotomy and broncheosphageal repair with symptomatic resolution and return to usual activities within 2 months.
We report a case of acquired bronchoesophageal fistula secondary to broncholithiasis. Such fistula is more commonly caused by infection, tumor, or trauma. Broncholithiasis is a rare disorder and usually the consequence of infection with tuberculosis, histoplasmosis, coccidiodides, actinomycosis or sarcoidosis with rare reports of silicosis as a cause[1,2]. We did not find any evidence of infection by tissue pathology or fungal serum titers and stains. However our patient came from Minnesota, a histoplasma-predominated area, and calcified mediastinal lymphadenopathy on CT is highly suggestive of and consistent with histoplasmosis infection. The most common symptoms of broncholithiasis are nonproductive cough often associated with hemoptysis. Fever or purulent sputum suggestive of respiratory infection is present 11-61% of the time[2,3]. Broncholithasis arises most commonly from the erosion and extrusion of calcified material from bronchopulmonary lymph nodes. In this case, a large lymph node was found between the esophagus and bronchus which eroded into the esophagus and caused the bonchoesophageal fistula. Importantly, the diagnosis of bronchioesophageal fistula is sometimes overlooked when the broncholith occupies the space between the bronchus and esophagus as occurred in our patient yielding the initial unremarkable gastrografin swallow study as described above. Bronchoscopy for broncholith extraction has been characterized as safe and effective for the removal of broncholiths, and the diagnosis of bronchoesophageal fistula was made only after bronchoscopic removal of the broncholith in this case.
Bronchoesophageal fistula is a rare disease potentially caused by broncholithiasis. The diagnosis may be missed if the broncholith occupies the space of the fistula. Clinical history can provide some diagnostic clues. Patients benefit from thoracotomy with bronchoesophageal repair.
Wei Peng, None.