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Recently described, the congenital Zika syndrome caused by the Zika virus has many features of other congenital infections. This case series study reports 22 infants with congenital Zika syndrome in Brazil who developed infantile spasms during their first year of life. The median age of infantile spasms onset was 4.3 (±1.4) months and ranged from 1 to 7 months. Neurologic evaluation showed that all 22 children presented severe impairment of neuropsychomotor development. A total of 18 infants had an interictal hypsarrhythmia electroencephalogram (EEG) pattern. Vigabatrin was prescribed as first-line treatment; however, only 5 patients were controlled. Steroid regimes were started for children who did not respond to vigabatrin and they are being followed. In conclusion, congenital Zika syndrome may be considered as an etiological agent of infantile spasms.