There are two previous reports of Candida dubliniensis endophthalmitis in North America. Here, we report a third case of C. dubliniensis endogenous endophthalmitis in a 31-year-old male patient who complained of left-sided decreased visual acuity. He had an associated mitral and tricuspid valve endocarditis, in the setting of intravenous drug use. Blood and sputum cultures were positive for C. dubliniensis. Fundoscopic examination was consistent with a fungal endophthalmitis. He was treated with fluconazole followed by intravenous liposomal amphotericin B for 6 weeks. C. dubliniensis is an important but rare cause of endophthalmitis in intravenous drug abusers.