The incidence of syphilis has greatly reduced in the severity of affected individuals due to the early treatment with antibiotics. However, due to the increase in the prevalence of human immunodeficiency virus (HIV) infection, it has also caused a concurrent rise in the number of neurosyphilis patients. Most common ocular manifestations include uveitis, interstitial keratitis, and vasculitis. A healthy 28-years old man presented with a progressive blurring of vision in the right eye for one month. It was his second episode. The first episode occurred 4 months back which resolved spontaneously without treatment. Vision in the right eye was 6/18. The pupil was dilated and the relative afferent pupillary defect was positive. Fundus examination showed a hyperemic swollen right optic disc with blurred margins and no macular fan or star. Vision, anterior segment, and fundus were normal in the left eye. After the investigations, he was diagnosed as a case of neurosyphilis with optic neuritis. He was treated with intravenous penicillin for two weeks followed by oral penicillin for three weeks. Vision in the right eye improved to 6/6; the optic disc swelling resolved with clear margins. He maintained good vision during the follow-up of six months. Early referral to an ophthalmologist upon suspicion of syphilis, detailed evaluation, and immediate treatment is mandatory to prevent permanent vision loss in these patients.
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