Purpose: We describe subtle macular changes that were detected by high-magnification color fundus imaging in infants with congenital Zika syndrome. Methods: This series included 4 infants (age: 1.4 ± 0.1 years [range, 1.3-1.5 years]) who were serologically confirmed with congenital Zika syndrome, examined under anesthesia, and underwent fundus examination and color fundus documentation. The fundus images were captured using 130° and 80° field lenses. Fluorescein angiography confirmed macular lesions. Results: Wide-angle color fundus images detected unilateral retinal findings in all 4 infants. Chorioretinal atrophy was identified in 2 eyes (25%) of 2 infants (50%) and pigment mottling in 3 eyes (37.5%) of 2 infants (50%). High-magnification imaging revealed subtle macular changes in the contralateral eyes of the 4 infants that were confirmed by fluorescein angiography imaging. Conclusions: High-magnification fundus imaging can be effective and reliable in revealing and documenting subtle structural changes in the macula of infants with congenital Zika syndrome.