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Bacteria can adhere to cardiac endothelium damaged by regurgitation or a shunt jet; however, healthy cardiac endothelium is supposedly resistant to bacterial adhesion. A 22-year-old man presented to our emergency department with fever. Physical examination revealed no obvious cardiac murmur, but there was evidence of splinter hemorrhages and Janeway lesions. Transthoracic echocardiography did not reveal vegetative lesions, but a 15 × 7-mm vegetation was identified on the surface of the left ventricular muscle just below the anterolateral commissure of the mitral valve without regurgitation or a shunt jet by means of transesophageal echocardiography. Surgery was performed on the seventh day, but the patient's postoperative course was unstable. Some complications occurred because the vegetation existed in a unique location. Although the patient continued to have an uncontrollable infection over the subsequent course, he was discharged on the 94(th) hospital day. We present a case of a vegetation in a unique location without exposure to regurgitation or a shunt jet. This case indicates that vegetative lesions may develop even in the absence of regurgitation and shunt jets. In case of infective endocarditis where a vegetation exists in a unique location, comprehensive testing or strategy are required to treat this condition.