We report the case of a 33-year-old patient with clinical history of echinococcosis admitted to our Hospital for the appearance of chest pain and electrocardiographic findings of anterior ischemia. The cardiac enzymogram was in the normal range, the chest roentgengram did not show any pathological findings, but two-dimensional echocardiography revealed the presence of a small circular area in the interventricular septum. Transesophageal echocardiography and cardiac nuclear magnetic resonance confirmed the presence of a small hydatid cyst in the middle ventricular septum; in addition, a myocardial scintigraphy revealed an apical stress defect with late reperfusion. Besides cardiologic therapy, the patient was treated with albendazole, an antiparasitic drug, 400 mg bid, for cycles of 28 days with 14 day withdrawal. After two cycles of albendazole therapy, two-dimensional echocardiography showed the absence of the round cystic mass of the interventricular septum previously described. In conclusion, in the case described, long-term therapy with albendazole determined the complete recovery from the illness with the simultaneous disappearance of the cyst and of clinical and electrocardiographic findings of myocardial ischemia.