Ocular syphilis can present with diverse manifestations. A 31-year-old male with chorioretinitis had positive VDRL and TPHA, with no genital lesions. Fundoscopy showed retinal lesions and macular edema. Patient was treated with intramuscular procaine penicillin 1.2 million units daily for 21 days and improved vision. This case emphasizes ocular syphilis can be the sole presenting sign and requires careful diagnosis. © 2013 Elsevier B.V., All rights reserved.