Background: Infective endocarditis (IE) is one of the most serious complications among patients undergoing hemodialysis. Mechanical disruption of the endothelium results in deposition of fi brin and platelet as a normal healing process. Such nonbacterial thrombotic endocarditis (NBTE) facilitates bacterial adherence and nurtures infected vegetations. Most common site of IE is mitral valve and aortic valve. We report an instructive case of IE with unusual site of vegetation at the left ventricular outfi ow tract. Case Report: 47-year old woman who is maintained hemodialysis was treated recurrent catheter related Methicillin-sensitive Staphylococcus aureus (MSSA) bacteremia. After three episodes of MSSA bacteremia and an episode of Candidemia, IE developed with vegetation at the left ventricular outfiow tract just above mitral valve. The patient received mitral valve replacement due to persistent infection despite proper antibiotics therapy and her septicemia got improved. Conclusion: IE of the left ventricular outfi ow tract is rare. Recurrent catheter related blood stream infection have contributed to damaging the LVOT endocardium, or the hemodynamic instability caused by hemodialysis and/or sepsis favored lesions of the endocardial surface initially, followed by the expression of chemokines, and fi nally the growth of vegetation.