Report a case of a male patient with a macular scar compatible with ocular toxoplasmosis (OT) in right eye (RE) and review the relevant literature on this disease. A patient, who attended for a routine contact lens follow up, presented with amblyopic exotropia without any ocular disease. Best-corrected visual acuity of the affected eye was 20/40 with constant and mono-fixation exotropia. Ophthalmoscopic assessment revealed a macular scar compatible with OT. OT is the leading cause of infection in the posterior segment. Inactive cases could be asymptomatic and diagnosis requires a complete eye examination, providing a correct diagnosis and patient management.