To report an unusual case of a patient with endogenous endophthalmitis caused by Actinomyces neuii.A 69-year-old woman in an immunosuppressed state and who had a previous history of periappendicular abscess presented with bilateral red painful eyes. The diagnosis was confirmed by culture and pan-bacterial polymerase chain reaction drawn from anterior chamber sample. On admission, the patient underwent an intravitreal injection of vancomycin combined with ceftazidime. Following a 3-week treatment of intravenous penicillin and topical sulfacetamide sodium, the patient recovered fully.Actinomyces neuii can cause endogenous endophthalmitis. Intravenous penicillin G is an effective treatment leading to favorable prognosis.