Aims/Purpose: Based on a clinical case seen in our setting, the purpose of this communication is to describe a case of ocular syphilis with an uncommon initial manifestation as well as its clinical and imaging features, diagnosis, treatment and evolution.Methods: We selected a case seen at the University Hospital Infanta Sofía in 2023. A 54‐year‐old male with Acquired Inmune Deficiency Syndrome (AIDS) under antiretroviral (ARV) treatment, diagnosed with cataract, who 1 month after surgery presented a paradoxical worsening of baseline visual acuity (VA) bilaterally accompanied by nyctalopia.Results: During the exam, we found VA from right eye (RE) of 0.03 and from left eye (LE) of 0.03. Fundus examination showed a bilateral central vitreous organization. Macular OCT from both eyes showed preserved foveal profile with pachychoroid associated with patchy absence of external limiting membrane, evident focal loss of ellipsoid zone and cysts in outer layers of the RE. Campimetry with practically abolished fields, neuroimaging study, complete serology including antirecoverin antibodies, and vitreous sample for microbiology were performed. From this study, it was found that the patient had a positive luetic seroconversion within a maximum period of five years. With these data, we oriented the differential diagnosis of the described retinopathy towards an infectious or toxic aetiology due to ARV drugs as the most probable causes. The patient was treated with intravenous penicillin with great improvement of vision and nyctalopia.Conclusions: Ocular syphilis is the great simulator par excellence and although infrequent, it can debut with atypical forms of uveitis.