Gastric antral vascular ectasia is an important cause of gastric bleeding. We herein report a case of gastric outlet syndrome due to a gastric antral vascular ectasia in a patient with a negative history for anemia or gastric bleeding. The patient underwent an antral resection. The specimen showed vascular ectasia, fibromuscular hyperplasia, and fibrin thrombosis, especially localized in the submucosa. The pathogenesis of this syndrome remains unclear. Surgery in cases of gastric outlet obstruction is the only reliable method to cure the patient. An antrectomy has been shown to be the procedure of choice. It carries a remarkably low rate of long-term sequelae and it can be performed in the majority of cases with low risk, especially in those patients not affected by cirrhosis or portal hypertension.