Purpose To present a case of bilateral endogenous endophthalmitis as the primary manifestation of an aortic root abscess and endocarditis, both of which were undetected by transesophageal echocardiogram. Design Observational case report. Methods A 13-year-old male presented with bilateral endogenous endophthalmitis and previously undiagnosed ventricular septal defect, subaortic stenosis, and a mitral valve cleft. Results Cardiac evaluation, systemic evaluation, and transesophageal echocardiogram were negative for endocarditis, but endocarditis and aortic root abscess were discovered at time of cardiac surgery. The patient responded to systemic treatment for endocarditis and surgical management of his cardiac defect. Conclusion A cardiac source for endogenous endophthalmitis should be considered in the presence of clinical diagnosis of bacteremia, despite a negative transesophageal echocardiogram and systemic evaluation.
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