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Purpose: To evaluate the surgical and functional outcomes in children with congenital Zika syndrome who underwent strabismus surgery., Methods: This prospective case series included children with congenital Zika syndrome who presented with horizontal infantile strabismus and underwent strabismus surgery. Ocular motility and visual fields were assessed preoperatively and postoperatively. Visual fields were considered normal if they exceeded 70 degrees in the temporal meridian. Postoperatively, parents and caregivers answered a questionnaire that assessed their child's behavioral improvements. A final ocular alignment within ±10 prism diopters (PD) and expansion of the visual fields were considered satisfactory motor surgical results at 6 months postoperatively., Results: Five children (3 girls [60%]) with congenital Zika syndrome (age: 36.4 ± 0.9 months) were included in this study. All children (100%) presented with moderate to severe visual impairment and 4 (80%) presented with funduscopic abnormalities. Preoperatively, 4 children (80%) had infantile esotropia (mean preoperative angle of deviation: 41.3 ± 6.3 PD) and 1 (20%) had infantile exotropia measuring 65 PD. The postoperative outcomes demonstrated ocular alignment in 4 children (80%) at the 6-month follow-up visit. Expansion of the temporal visual field was observed in 7 eyes (70%) of 4 children (80%). Four caregivers (80%) reported behavioral improvement in daily activities, and all caregivers (100%) reported improved peripheral target detection and socialization skills., Conclusions: Strabismus surgery can be an effective procedure for treating horizontal strabismus in children with congenital Zika syndrome because it can improve ocular alignment, expand the visual field, and improve the child's social, functional, and behavioral skills. [J Pediatr Ophthalmol Strabismus. 2020;57(3):169-175.]., (Copyright 2020, SLACK Incorporated.)