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To describe the ocular findings of two infants with congenital Zika syndrome born in São Paulo state, Brazil, outside the primary outbreak region.Two female infants with congenital Zika syndrome were submitted to a complete screening for infectious diseases, as well as ocular examination, fundus photography, and optical coherence tomography of the retina. One of the infants was submitted to ocular ultrasound.Both children showed positive serology for Zika virus, and other infectious diseases were ruled out. On fundus examination, one newborn had extensive bilateral macular chorioretinal atrophy with hyperpigmented borders associated with gross pigment mottling, as well as atrophic well-limited roundish peripheral lesions in the right eye. Spectral domain optical coherence tomography of the right eye showed complete chorioretinal atrophy in the temporal quadrant. The other infant presented bilateral gross pigment mottling in a paramacular location and a normal optical coherence tomography.In this report, the ophthalmologic findings of two infants with Zika syndrome and ocular findings born in São Paulo state, Brazil, are described. Optical coherence tomography findings demonstrate the broad range of retinal damage caused by congenital Zika virus infection.The autochthonous cases of infants with congenital Zika syndrome and ocular findings described in this report demonstrate that Zika virus infection is not limited to northeast Brazil, with the potential for even greater spread.