A 42-year-old man experienced abrupt reduction of vision in the left eye and had vitreous opacity and hypopyon on initial examination. He underwent an emergency vitrectomy and phacoemulsification with posterior capsulectomy, followed by a 2-week course of intravenous fluconazole and antibiotics. The final visual acuity was 20/20 OS, Amoebas, together with polymorphonuclear cells, were observed on examination of specimens of the aqueous humor and vitreous fluid obtained during the surgery. Staphylococcus epidermidis was also cultured from the vitreous fluid. The amoebas had a crescent-shaped periphery of the cytoplasm and ambiguous nuclei. Ultrastructurally, the cytoplasm was surrounded by a thick electron-dense coating. The early vitrectomy and combined use of antibiotics and antifungals might have led to the good visual outcome in this patient.