Cytomegalovirus-Associated Esophagopulmonary Fistula in an AIDS Patient Successfully Treated with Esophageal Stenting

Abstract

Introduction: A 46-year-old man with AIDS and poor antiretroviral adherence presented with a 12-day history of chest pain, dyspnea, cough, and fever. Examination revealed tachycardia, cachexia, and thrush. One month prior, CD4 count was 9/mm3 and HIV viral load was 57,647 copies/mL. Chest CT noted a dominant cavitary mass in the right upper lobe, several smaller pulmonary cavitations, and proximal esophageal dilatation. Treatment for bacterial pneumonia and candidiasis was started. Initial infectious studies, including acid-fast sputum smears, were negative. Despite 7 days of broad-spectrum antibiotics, his cough worsened, serial chest imaging noted disease progression, and sputum culture grew MRSA and commensal bacteria. Three weeks into his course, he developed dysphagia and odynophagia. Serum PCR for Cytomegalovirus (CMV) was 701 copies/mL. EGD noted a cratered 5-cm longitudinal ulcer in the proximal esophagus (Figure 1A) and a 1-mm fistulous opening within the ulcer (Figure 1B). Barium esophagram revealed a communication between the proximal esophagus and right hemithorax (Figure 1C). Chest CT demonstrated tracking of barium from the esophagus into the dominant pulmonary cavity (Figure 1D). These findings were consistent with the diagnosis of an esophagopulmonary fistula. On repeat EGD, biopsies of the ulcer were obtained, followed by placement of a 12-cm fully-covered metal stent across the fistula. Pathology showed esophageal squamous epithelium, inflamed reactive stroma, and acutely inflamed columnar respiratory-type mucosa with scattered CMV-positive cells, confirmed on immunohistochemistry (Figure 1E). The patient received valgancyclovir for 21 days and the stent was removed 35 days after placement with resolution of the ulcer. Repeat barium esophagram showed no structural abnormalities. We present the first reported case of a CMV-associated esophagopulmonary fistula successfully treated with antiviral therapy and a fully-covered metal esophageal stent. Esophagorespiratory fistulas are rare and usually associated with malignancy or post-operative complications. Use of fully-covered metal stents has been described in the treatment of esophageal fistulas and leaks with variable success.Figure 1