Actinobacillus actinomycetemcomitans is a rare cause of infection in humans. It has been mainly reported in cases of endocarditis and destructive periodontal disease. This report describes the case of a 51-year-old female who had an acute onset of anterior iridocyclitis, followed by the appearance of a white nodule on the lens and subsequent involvement of the vitreous. The course of the disease was long and insidious. Actinobacillus actinomycetemcomitans was isolated from 3 of 26 blood cultures drawn over a period of 40 days, and also from the vitreous. The patient had been suffering from periodontal disease for 3 months prior to her admission. Only two cases of endophthalmitis presumably caused by this organism have previously been reported, but the organism has never been recovered from the eye. This report presents the first case of culture-proven Actinobacillus actinomycetemcomitans endophthalmitis following destructive periodontal disease.