To report a case of a neurosyphilis patient with cilioretinal artery occlusion as the first manifestation.

A 58-year old man presented with a visual impairment which developed 5 days earlier in his right eye. During initial evaluation, the visual acuity in the right eye was 0.02. On fundus examination, a whitening adjacent to the macula was observed and fluorescein angiography showed filling delay and late leakage of the cilioretinal arteries. The patient was diagnosed with cilioretinal artery occlusion. Two weeks after his initial visit, visual acuity in the patient's right eye improved to 0.7 and retinal whitening was decreased on fundus examination. Three months later, a visual decrease in the right eye reoccurred and the visual acuity was 0.01. Anterior chamber cell reaction with hypopyon was observed on slit lamp examination and vitreal opacity was noted during fundus examination. Among the numerous tests performed in order to determine the cause of uveitis, RPR antibody and treponemal antibody were found positive. The patient was diagnosed with neurosyphylis and 2 weeks after intravenous administration of penicillin, visual acuity improved to 0.7 and anterior chamber cell reaction as well as vitreal opacity decreased.

The authors reported a case of cilioretinal artery occlusion suspected secondary to neurosyphilis.