QJM Advance Access published August 26, 2012

Q J Med doi:10.1093/qjmed/hcs170

Clinical picture
Bronchoesophageal Fistula Secondary to Broncholith
An 85-year-old African-American man with past medical history significant for hypertension, diabetes mellitus, COPD, remote pulmonary tuberculosis (TB) and lung silicosis, presented with 1 month history of dyspnea, fever, dysphagia and chronic cough with purulent sputum. TB was treated with quadruple therapy 15 years ago and has not had respiratory symptoms since. His occupation for most of his lifetime was construction and demolition. He smoked 1 pack of cigarettes daily for 50 years and quit 10 years ago. Examination of the chest revealed bilateral expiratory wheezing more prominent on the right side with dullness to percussion on the right base. A chest roentgenogram (CXR) showed right lower lobe consolidation suggesting pneumonia. Given his past history of TB and current dysphagia, a computed tomography (CT) of the chest was done, which showed right lower lobe consolidation, and multiple hilar and mediastinal calcifications with a bronchoesophageal fistula coursing between the right bronchus intermedius and the esophagus (Figure 1, arrow). Furthermore, a broncholith was noted in the lumen of the fistula (Figure 1, arrow head). The patient was started on intravenous antibiotics and oral intake was stopped to prevent further aspiration. An esophagogastroduodenoscopy (EGD) was performed and the bronchoesophageal fistula was identified at 26 cm from the incisors. At the opening of the fistula there was a broncholith obstructing the fistula (Figure 2, arrow). A stent was successfully placed over the guide wire covering the fistula. The patient was started on enteral feeding through a percutaneous endoscopic gastrostomy (PEG) tube to minimize risk of recurrence of aspiration pneumonia. He was discharged in stable condition.

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Discussion
Broncholithiasis is a rare disorder characterized by parenchymal and hilar calcifications in response to a chronic inflammatory process.1-3 Bronchopulmonary fistula caused by broncholithiasis is extremely rare. It occurs mostly in the right side of the bronchial tree.4 The most common clinical features include: chronic cough with or without sputum production, lithoptysis, dysphagia, wheezing, hemoptysis, fever and chills. The diagnosis can be

Figure 1. CT chest showed presence of a bronchoesophageal fistula coursing between the bronchus intermedius and the esophagus (arrow). A broncholith was noted in the lumen of the fistula (arrow head).

Figure 2. EGD was able to identify the bronchoesophageal fistula at 26 cm from the incisors. A broncholith was seen at the opening of the fistula.

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Clinical picture challenging in cases of atypical presentation as it can involve different locations within the bronchial tree, as well as the possible involvement of other mediastinal structures.5 Erosion into major vascular structures have been reported with devastating consequences. The management of broncholithiasis is determined by the size as well as degree of attachment of the broncholith to the bronchial wall.5 Removal of large broncholiths can be achieved with rigid bronchoscopy with reported success rates from 67% to 87%.1,4 However, perforation and hemorrhage especially when laser is utilized to fragment large stone have been reported with this method.1,6 Removal of firmly attached broncholith to the bronchial wall is associated with significant hemorrhage, air leak and empyema.5,6 Surgical removal as well as esophageal stenting have been used for symptomatic treatment and closure of fistulous tracts.7 In this case, giving the large broncholith size and the patient advanced age, surgery was deferred by the patient and his family, and he underwent esophageal stent placement with PEG tube for feeding. Chronic cough, recurrent lung infections and dysphagia, should always raise the suspicion for bronchoesophageal fistula which warrant carefully evaluated with advanced imaging and endoscopic procedures. Photographs and text from: A.H. Alraiyes, Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA, USA; R. Desai, Cleveland Clinic Lerner College of Medicine of Case Western Reserve University Cleveland Clinic, Cleveland Clinic, Cleveland, OH, USA; M. Auron, Department of Hospital Medicine, Cleveland Clinic Lerner College of Medicine of Case Western Reserve University, Cleveland Clinic, Cleveland, OH, USA; M.D. Castillo, Pulmonary Critical Care Department, Case Western Reserve University (Metrohearlth), Cleveland, OH, USA; M.C. Alraies, Department of Hospital Medicine, Cleveland Clinic Lerner College of Medicine of Case Western Reserve University, Cleveland Clinic, 9500 Euclid Avenue, A13, Cleveland, OH 44195, USA. email: alraiec@ccf.org Conflict of interest: None declared.

References
1. Ferguson JS, Rippentrop JM, Fallon B, Ross AF, McLennan G. Management of obstructing pulmonary broncholithiasis with three-dimensional imaging and holmium laser lithotripsy. Chest 2006; 90912. 2. Choudhary C, Gildea TR, Salman R, Guzman ED, Mehta AC. Management of tracheomediastinal fistula using selfexpanding metallic stents. Ann Thorac Surg 2008; 85:18002. 3. Shaaban AM, Mann H, Morrell G, Carveth H, Elstad MR. A case of broncholithiasis and esophagobronchial fistula. J Thorac Imaging 2007; 22:25962. 4. Go T, Kobayashi H, Takata M, Shirasaki H, Miyayama S. Endoscopic management for broncholithiasis with bronchoesophageal fistula. Ann Thorac Surg 2007; 84:20935. 5. Olson EJ, Utz JP, Prakash UB. Therapeutic bronchoscopy in broncholithiasis. Am J Respir Crit Care Med 1999; 160:76670. 6. Menivale F, Deslee G, Vallerand H, Toubas O, Delepine G, Guillou PJ, et al. Therapeutic management of broncholithiasis. Ann Thorac Surg 2005; 79:17746. 7. van den Bongard HJ, Boot H, Baas P, Taal BG. The role of parallel stent insertion in patients with esophagorespiratory fistulas. Gastrointest Endosc 2002; 55:1105.

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