We report a case of cystoid macular edema (CME) secondary to syphilitic uveitis that was successfully treated with pars plana vitrectomy with internal limiting membrane peeling. A 37-year-old male with a history of HIV developed a CME secondary to syphilitic panuveitis. His uveitis resolved following treatment with intravenous penicillin, yet his CME persisted and was refractory to four posterior sub-tenon triamcinolone acetonide injections. A pars plana vitrectomy with internal limiting membrane peeling was performed, resulting in lasting resolution of the CME and the improvement of his visual acuity at the two-month follow-up visit. Pars plana vitrectomy with internal limiting membrane peeling may be a viable alternative for the treatment of CME in patients with syphilitic uveitis. In particular, it may serve as a viable alternative for the treatment of CME in patients with a history of infectious uveitis or other comorbidities, such as HIV infection.