Purpose: To describe the presentation and clinical course of a transgender patient with central serous chorioretinopathy (CSCR) and discuss the role of exogenous testosterone use in CSCR in this patient population. Method: Case report. Results: A 63-year-old transgender male with a history of exogenous testosterone use for over twenty years was referred to retina clinic for evaluation of macular pathology. The patient’s most recent total testosterone levels prior to presentation were 962 and 1,179 ng/dL (normal cisgender male levels for age 60+: 300-720 ng/dL). Fundus imaging revealed subretinal fluid involving the fovea without choroidal neovascularization in the right eye. There was no recent corticosteroid use. The patient’s dose of testosterone was reduced. After four months of observation, the subretinal fluid persisted and was successfully treated with half-fluence photodynamic therapy (PDT). At last follow-up, 15 months after PDT, the patient remained stable without recurrence and the best-corrected visual acuity was at baseline. Conclusion: Exogenous testosterone use with elevated serum testosterone levels may be associated with the development of central serous chorioretinopathy in the transgender patient population. CSCR in these patients might be effectively treated with PDT and testosterone dose reduction. Ophthalmologists should be aware of potential side effects of testosterone supplementation in this patient population, as well as the role of testosterone in CSCR pathophysiology.