The most common cause of esophagorespiratory fistulas (ERFs) is associated with malignancy. The use of self-expandable metal stents is effective for the treatment of malignant ERFs, but benign ERF is rare, which is why its optimal treatment is not defined yet. There have been few reports describing benign esophagopleural fistula and its treatments in South Korea. Here, we report a rare case of spontaneous esophagopleural fistula, which was successfully treated by endoscopic placement of a membrane covered metal stent.
Esophagorespiratory fistula (ERF) is an uncommon condition, despite of the anatomical proximity of these structures. Malignancy of esophagus, lung, or mediastinum is recognized as the most common cause. Causes of ERF development include direct tumor invasion and subsequent perforation or after radiation, laser therapy, chemotherapy, or pre-existing stents.
A 73-year-old man was admitted to Chungnam National University Hospital complaining right chest discomfort. He had 20 pack-years smoking history but had neither an operation history nor a medical history such as diabetes, hypertension, pulmonary tuberculosis, or hepatitis. Physical examination was unremarkable except for diminished respiratory sounds in the whole right lung field. The results of laboratory studies showed a white blood cell (WBC) count of 8,500/mm2; hemoglobin of 12.5 g/dL; and platelet count of 164,000/mm2. The serum biochemistry presented Na+ of 140 mEq/L; K+ 4.3 mEq/L; Cl- 107 mEq/L; aspartate aminotransferase 19.5 IU/L; alanine aminotransferase 12.4 IU/L; total bilirubin 0.9 mg/dL; and lactate dehydrogenase (LDH) 246 IU/L (all within normal ranges) with hypoalbuminemia (2.8 g/dL) and elevated serum creatinine level (1.65 mg/dL). Chest X-ray revealed right pleural effusion (
ERF is used to describe all fistulas located between the esophagus and the airway tree; however, esophagotracheal and esophagobronchial fistulas mostly account for the remaining ERF cases (3% to 11%) of esophagopulmonary fistulas.
Known causes of acquired benign ERF include trauma and infection.
Esophagopleural fistulas rarely heal spontaneously. Leaks of the esophagus are associated with a high mortality rate and need to be treated as soon as possible. Therapeutic options include surgical repair or resection or conservative management with antibiotic therapy and cessation of oral intake. Surgical correction requires aggressive and technically demanding procedures, results for which are uncertain. Endoscopic techniques to overcome this issue have been reported with obliterating agents, such as fibrin glue or cyan acrylic glue,
Self-expanding metal stents (SEMSs) have been considered a safe and effective treatment for malignant dysphagia stricture, and fistula in inoperable patients.
In our case, the cause of the esophagopleural fistula was unclear. However, esophagopleural fistula was successfully treated by an endoscopic stent insertion. Therefore, it appears that implantation of membrane-covered metal stents is an effective alternative for the treatment of esophagopleural fistula.
The authors have no financial conflicts of interest.
Chest X-ray showing (A) a huge left pleural effusion and (B) inserted chest tube catheter.
Esophagogram showing apparent leakage from above the esophagogastric junction to the left pleural cavity.
Endoscopy images showing (A) a purulent fistular tract opening in the lower esophagus and (B) fully expanded stent.
Endoscopy images showing (A) the lower esophagus after stent removal and (B) complete closure of the esophagopleural fistula 6 months later.