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A patient presenting with signs and symptoms suggesting myocardial ischemia was found to have anomalous origin of his right coronary artery from the left aortic sinus of valsalva associated with a bicuspid aortic valve. Abnormal left ventricular function with anaerobic myocardial metabolism was documented in the absence of significant coronary artery disease or aortic obstruction. The possible relationship of these findings to the coronary artery anomaly and bicuspid aortic valve is considered. The necessity of identification of the origin of the coronary arteries is re-emphasized in the evaluation of patients with bicuspid aortic valves coming to cardiac catheterization. Anomalous origin of a coronary artery is a rare occurrence. The association of this anomaly with a bicuspid aortic valve has not been emphasized and the possible clinical consequences of this combination of malformations have not been described. We recently evaluated a patient with anomalous origin of right coronary artery from the left coronary ostium and a bicuspid aortic valve. A description of these findings follows.