A 39-year-old male with a history of cardiac murmur from early childhood was diagnosed as having infectious endocarditis (IE) complicated by discrete subaortic stenosis (DSS). Echocardiography revealed severe aortic regurgitation, subaortic membranous structure and mild mitral regurgitation. The pressure gradient across the subaortic stenosis was 105 mmHg according to continuous Doppler wave ultrasonography. Aortic valve replacement and resection of subaortic membranous tissue and mitral annuloplasty were performed. Postoperative cardiac catheterization demonstrated that the hemodynamic data were remarkably improved, and the patient was free of symptoms. He is currently well at 6 months after the operation.