Aortico-left ventricular tunnel (ALVT) is a rare congenital anomaly presenting abnormal connection between the ascending aorta and the left ventricle. In most reported cases, the aortic end of the tunnel is above the right coronary sinus. Cases of ALVT related to the left aortic sinus are extremely rare. We herein report a case diagnosed preoperatively as ALVT arising from the left aortic sinus. The actual diagnosis observed at surgery was aortic valve insufficiency with a left ventricular outflow tract aneurysm. We successfully performed aortic valve repair and plication of the left ventricular aneurysm.
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