AbstractPurpose: To describe the clinical and laboratory findings, ocular complications, and visual prognosis of patients with syphilis‐associated uveitis.Methods: Retrospective observational case series of patients diagnosed with ocular syphilis between January 2012 and April 2022 at a single tertiary referral centre in Portugal. Medical records of patients presenting with syphilis‐associated uveitis were reviewed.Results: Eighteen eyes from 10 consecutive patients were included. All patients were male and median age at diagnosis was 51 years (range 20–83). HIV coinfection was present in 3 patients, with two of these being newly diagnosed on presentation. All patients had a reactive serum treponema pallidum haemagglutination test and median serum rapid plasma reagin titre was 1:260 (range 1:1–1:520), with no significant difference between HIV‐positive and HIV‐negative patients (p = 0.222). A lumbar puncture was performed in seven patients, confirming the diagnosis of neurosyphilis in six patients. Bilateral involvement occurred in eight patients. The most frequent form of intraocular inflammation was posterior uveitis (33.3%), followed by panuveitis (27.8%) and anterior uveitis (27.8%). All patients were treated with intravenous penicillin, showing partial or complete resolution of the ocular findings. Visual acuity improved significantly from presentation to the last visit (0.7 logMAR, range 0–2; and 0.2 logMAR, range 0–0.7, respectively; p = 0.004). Five eyes (27.8%) had ocular complications from syphilis‐associated uveitis, including epiretinal membrane formation and cystoid macular oedema, with significantly worse visual prognosis (p < 0.001).Conclusions: Ocular syphilis is re‐emerging as a prominent cause of uveitis. In our series, patients were characteristically middle‐aged men with posterior segment involvement. A relevant proportion of patients had HIV co‐infection. Despite good visual prognosis with standard treatment, ocular complications may compromise visual recovery.