Ann Thorac Surg 2002;73:302-304
© 2002 The Society of Thoracic Surgeons
Case report
Perforating Barrett’s ulcer resulting in a life-threatening esophagobronchial fistula
John J. Nigro, MD, MS*a,
Ross M. Bremner, MD, PhDa,
Clark B. Fuller, MDa,
Jörg Theisen, MDb,
Yanling Ma, MDc,
Vaughn A. Starnes, MDa
a Department of Cardiothoracic Surgery, University of Southern California Keck School of Medicine, Los Angeles California, USA
b Department of General Surgery, University of Southern California Keck School of Medicine, Los Angeles, California, USA
c Department of Pathology, University of Southern California Keck School of Medicine, Los Angeles California, USA
Accepted for publication April 5, 2001.
* Address reprint requests to Dr Nigro, Department of Cardiothoracic Surgery, University of Southern California Keck School of Medicine, 1510 San Pablo St, Suite 415, Los Angeles, CA 90033, USA
e-mail: jnigro{at}hsc.usc.edu
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Abstract
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Perforating benign ulcer is a very rare complication of Barrett’s esophagus. This report presents the management of a patient with a Barrett’s ulcer that penetrated into the left mainstem bronchus resulting in a life-threatening bronchial esophageal fistula. This rare complication was successfully managed by using a staged surgical approach, which combined the principles used for treating benign esophagorespiratory fistulas and perforating Barrett’s ulcers.
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Introduction
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Benign peptic ulceration within Barrett’s epithelium occurs in approximately 10% to 24% of patients with Barrett’s esophagus [1]. Perforation of these ulcers is a rare but life-threatening complication that has a mortality of close to 50% [1]. Penetration can occur into any adjacent structure [1–3], but a bronchoesophageal fistula due to a perforating Barrett’s ulcer has been reported only once previously [4]. This report details the definitive surgical management of a patient with a Barrett’s ulcer that eroded into the left main stem bronchus resulting in a bronchoesophageal fistula.
A 77-year-old man with a history of heartburn and esophageal stricture experienced a sudden onset (paroxysm) of intense coughing and vomiting. Initial workup revealed a left lower lobe pneumonia with a large fistula between his esophagus and airway. Endoscopy confirmed the presence of a large fistula, but there was no evidence of malignancy and a combination gastrostomy/feeding jejunostomy tube was placed to allow for both enteral feeding and diversion of gastric secretions. The patient was then transferred home but was readmitted with recurrent pneumonia and referred to the Department of Cardiothoracic Surgery. Since the original coughing episode, the patient had lost 40 lbs. He was febrile, had an elevated white blood count (14,000 cells/hpf), had renal dysfunction (serum creatinine, 2.4 mg/dL), and had a low serum albumin level (2.2 mg/dL). A computerized tomographic scan localized the communication to the left main stem bronchus (Fig 1)
and revealed a left lower lobe pneumonia with a parapneumonic fluid collection. No evidence of malignancy was identified. Subsequently, combined esophagoscopy and bronchoscopy confirmed the presence of a large communication between the esophagus and left main stem bronchus with "clean punched-out" borders (Fig 2)
and a hiatal hernia. No esophageal mass or other evidence of neoplasia was identified. Barrett’s changes were present up to the midesophagus, and four quadrant biopsies from each 2 cm of the Barrett’s segment were obtained; additional biopsies were obtained at the fistula site. Esophageal diversion was accomplished with a cervical esophagostomy. After diversion the septic state resolved and the patient was discharged home with tube feedings and high-dose acid suppression therapy. Pathology results confirmed the presence of Barrett’s metaplasia from the gastroesophageal junction up to 27 cm (from the incisors). He was readmitted 6 weeks later for esophageal resection and reconstruction of his alimentary tract. A right posterolateral thoracotomy was performed, the esophagus mobilized, and the fistula was approached through the esophagus. The defect in the bronchus was closed with an intercostal muscle flap patch, placing the pleural surface of the muscle patch on the luminal surface of the bronchus. Chest tubes were placed in the standard fashion and the thoracotomy incision was closed. After positioning the patient in the supine position, an upper midline abdominal incision was completed and the stomach mobilized. Continuity was reestablished by placing the stomach in the retrosternal position and by performing a cervical esophagogastrostomy. The resected esophagus is seen in Figure 3;
it had Barrett’s metaplasia, mucosal erosion with ulceration, and a 1-cm2 transmural defect (fistula). Histology confirmed the presence of inflammation and intestinal metaplasia (Fig 4).
The postoperative course was complicated by a left lower lobe pneumonia, but the patient was successfully extubated on postoperative day 4. A barium esophogram revealed an intact anastomosis with easy flow of contrast through the stomach; the patient subsequently began oral feedings, and was transferred home on postoperative day 13. On follow-up bronchoscopy there was healing of the bronchial fistula/patch site without stricture or granulation tissue formation.
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Fig 1. Soft tissue windows of the chest computed tomographic scan. The esophagobronchial fistula is marked with arrow A. Arrow B identifies the air-filled and dilated esophagus.
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Fig 2. Endoscopic view of the distal esophagus, the large black arrow points to the fistula between the esophagus and the left mainstem bronchus. The picture on the right shows both the fistula and the esophageal lumen. Endoscopic Barrett’s changes are evident in both photographs.
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Fig 3. Resected esophagus that has been incised longitudinally, exposing the mucosal surface. The distal esophagus is marked with "D" and the fistula site is marked with "F." The white mucosa at the superior aspect of the esophagus is squamous mucosa, the area adjacent to the fistula has severe mucosal damage, and the distal esophagus has salmon pink mucosa characteristic of Barrett’s esophagus.
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Fig 4. Sections from the resected esophagus that have been stained with hemotoxilyn and eosin. (A) Section of esophageal wall at the fistula site that contains inflammation, fibrosis, and erosion. (B) Esophageal mucosa adjacent to the fistula. It contains intestinal metaplasia with goblet cells (Barrett’s esophagus).
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Comment
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Perforating ulcer is a rare but life-threatening complication of Barrett’s esophagus [1–3], it is a benign transmural erosion located within metaplastic epithelium, a recent review of both the English and French literature identified only 31 previous reports [1]. The overall mortality in this series was 45% and an aggressive surgical approach with early esophagectomy was advocated [1]. Penetration into the tracheobronchial tree occurred in 4 patients [1]. This communication between the esophagus and the airway constitutes a rare type of benign-acquired esophagorespiratory fistula. Management of these fistula is surgical with isolated reports of successful medical management [5, 6]. The principles for successful fistula management are: (1) control of sepsis, (2) establishment of a good nutritional status, (3) pulmonary support and rehabilitation, and (4) surgical repair or bypass [4, 6–12]. Despite gastric drainage, jejunal feedings, and broad-spectrum antibiotic therapy, soiling of this patient’s bronchial tree continued and control of sepsis was not obtained. Because of his poor nutritional status, active infection, and renal dysfunction, his operation was completed in two stages. Esophageal diversion was performed with a cervical esophagostomy and gastric secretion was controlled with high-dose proton pump inhibitor therapy. Resection and reconstruction was undertaken after 6 weeks of enteral (jejunal) nutrition and antibiotics. Although bypass and exclusion of this type of fistula have been previously reported [1, 4], esophagectomy was performed. The bronchial defect in this patient was approached through the esophagus to limit enlargement of the defect. Reconstruction was performed through the retrosternal route, avoiding interference with the bronchial repair and to limit the possibility of fistula recurrence. The patient’s stomach was used for this reconstruction; alternatively, a substernal colon interposition could have been used. Finally, a tube jejunostomy was placed to secure the ability to provide enteral feedings.
This report presents the management of a patient with Barrett’s ulceration into the left main stem bronchus. We believe this is a severe complication of the relatively common disease of gastroesophageal reflux and that timely antireflux operation may have prevented the development of this life-threatening lesion. However, this fistula was successfully managed by combining the principles used for the treatment of benign communications between the esophagus and the airway with those advocated for the management of Barrett’s perforating ulcers.
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References
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Abstract
Introduction
