Abstract
PURPOSE: To report an unusual case of congenital bilateral abnormality due to coexisting ocular toxoplasmosis and retinopathy of prematurity. OBSERVATIONS: A prematurely born baby with bilateral retinopathy of prematurity (ROP) (one eye treated) was found to have a hyperpigmented torpedo maculopathy lesion in one eye while the other eye presented with microphthalmia, congenital cataract and non-specific pigmentary retinopathy. Following negative TORCH screening, laboratory tests subsequently revealed increased plasma IgG and IgM for toxoplasmosis. CONCLUSIONS: We present an unusual case of coexistence of congenital ocular toxoplasmosis with torpedo maculopathy and retinopathy of prematurity.