An unusual cause of upper gastrointestinal bleeding

Abstract

A 46-year-old man with stage IV esophageal cancer (Figure 1, left) presented with dysphagia and weight loss. A palliative esophageal stent (ultraflex) was placed to relieve the dysphagia (Figure 1, right) in another hospital for symptom relief. He was administered concurrent chemoradiotherapy after oral intake resumed. On the third hospital day of the fourth chemotherapy course, he experienced sudden-onset large-volume hematemesis and went into shock. After resuscitation, an emergency endoscopy demonstrated bloody content in the esophagus, and the bleeding was uncontrollable. A contrast-enhanced chest computed tomography (CT) was performed (Figure 2). What is the diagnosis? The chest CT demonstrated active contrast extravasation from the aorta (Figure 2, left) into the upper margin of the esophageal stent (Figure 2, right), and aortoesophageal fistula (AEF) was diagnosed. The patient declined surgery because of advanced esophageal cancer. Emergency endovascular repair using an aortic stent was performed to control the bleeding. The patient experienced severe pneumonia after resolution of the bleeding episode, and a tracheoesophageal fistula was found. A week later, he died of multiple organ failure. Esophageal stent for advanced esophageal cancer is aimed at maintaining oral intake to improve the quality of life.1-3 Stent-related major complications, such as perforation and hemorrhage, occur more frequently after radiotherapy and limit its use as a bridge to surgery or before preoperative chemoradiotherapy.1 Patients with AEF may experience mid-thoracic pain, heralded hemorrhage, and then fatal hemorrhage due to rapid blood extravasation from the aorta to the esophagus, so-called Chiari's triad.4 Unlike other causes of upper gastrointestinal bleeding, AEF can be diagnosed using endoscopy in less than 10% of cases. Diagnosis is usually made using CT arteriogram showing active extravasation of blood from the aorta to the esophagus. The treatment options for AEF include definitive surgical aortic replacement, temporary endovascular aortic repair, or further esophageal stenting. The prognosis is poor because most of these patients have contraindications to major surgery due to poor general condition. The authors receive funding from Changhua Christian Hospital 106-CCHIRP-030 for this manuscript. The authors declare no conflict of interest. Yu-Chun Hsu performed the endoscopy and final approval of the manuscript. Chia-Bung Chen provided 3-D reconstruction of the image and final approval of the manuscript. Hsu-Heng Yen was involved in drafting, writing and final approval and drafting of the manuscript.