A rare cause of esophagopleural fistula due to intensity-modulated proton therapy: a case report and review of literature

Abstract

Esophagopleural fistula (EPF), initially described in 1960, is an abnormal communication between the esophagus and the pleural cavity which can occur due to congenital malformation or acquired due to malignancy or iatrogenic treatment. The most common presenting symptoms are of a respiratory infection, such as fever, chest tenderness, cough and imaging findings consistent with pleural fluid consolidation. In this report, we present a 59-year-old man who exhibited shortness of breath, productive cough, and significant weight loss for 2 weeks. His medical history was significant for smoking-related lung disease and pulmonary squamous cell carcinoma (SCC). His SCC (T4N0) was diagnosed 6 years prior to this presentation and was treated with chemoradiotherapy. The cancer recurred a year ago and he was treated with intensity-modulated proton therapy (IMPT) and consolidation chemotherapy. During admission, he was found to have an EPF by CT scan after initially failing antibiotic treatment for suspected complicated pneumonia and pleural effusion. Multiple attempts of esophagopleural fistula closure were made using endoscopic self-expandable metallic stents and placement of an esophageal vacuum-assisted closure device. However, these measures ultimately failed and, therefore, he required an iliocostalis muscle flap (Clagett window) procedure for closure. Esophageal pulmonary fistulae should be suspected whenever patients have undergone thoracic IMPT and may present with acute pulmonary complications, particularly pneumonia refractory to antibiotic treatment. This case reviews the current literature, potential complications, and treatment options for esophagopleural fistulas.

Keywords: Chemoradiation; Endoscopy; Esophageal stent; Esophagopleural fistula; Pleural effusion.

Figure 1.

CTA chest demonstrating right middle (A) to lower lobe (B) cavitating consolidation with loculated pleural effusion

Figure 2.

CT chest demonstrating air-fluid level in the right basilar pleural effusion and unchanged consolidation in the right lung

Figure 3.

CT chest demonstrating a fistulous connection between the lower esophagus with the air and debris filled right-sided pleural cavity

Figure 4.

Endoscopic placement of self-expandable metallic stent (SEMS) in the middle third of the esophagus for esophago-pleural fistula

Figure 5.

Endoscopic placement of esophageal vacuum-assisted closure (EVAC) for persistent esophago-pleural fistula

Figure 6.

Esophagogram showing contrast extending outside of the proximal margin of the stent on the left, pooling between the stent and esophageal wall. No extravasation outside the esophageal lumen, demonstrating the closure of esophago-pleural fistula after surgical repair with muscle flap