AbstractObjectiveTo describe oral alterations in children with congenital Zika syndrome (CZS).MethodsThis was a case series, whose research instrument was a structured questionnaire, associated with the use of medical record data and extra and intraoral clinical examination.ResultsThirty‐two children were evaluated, the majority male (18/32%–56.3%), mean age 22 months (SD = 2.71). It was also observed that the majority of the patients (19/32%–59.4%) presented a low family income. All the children had a mean head circumference of 29.43 cm (SD = 1.42). Regarding the alterations, an ogival‐shaped palate was observed in 14 children (43.7%), and delayed chronology of eruption was observed in 15 children (46.9%), of whom 7 children (21.9%) did not present eruption of the upper left lateral incisor (p = .0002) and upper right lateral incisor (p = .002) until the moment of analysis. Additionally, 03 children with yellowish dental pigmentation were identified in erupted teeth after the onset of phenobarbital use. Enamel hypoplasia was identified in 9 children (28.1%) and only one child with ankyloglossia.ConclusionCZS may present delayed chronology of eruption, ankyloglossia, ogival‐shaped palate, and enamel hypoplasia, requiring dental follow‐up aimed at prevention, promotion, and rehabilitation of the health of these children.