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Dental enamel defects (DED) are lesions that occur due several factors. Proper care is needed to promote their treatment and prevention. The aim of this study was to evaluate the occurrence of DED in permanent teeth of children who used antimicrobial drugs in the first four years of life. This is a crosssectional study carried out in a Primary Health Care (PHC) service, which included children from six to 12 years of age. DED were evaluated by oral examination, and data on the use of antimicrobials in early childhood were collected based on medical records. Data were analyzed with the chi-square test and Fisher's exact test. The sample included 144 children. In relation to DED, 50% (72) and 20.1% (29) presented opacity and hypoplasia, respectively. Amoxicillin was the most frequently prescribed drug, followed by sulfamethoxazole + trimethoprim. Among the children, 78.5% (113) were prescribed antimicrobial drugs at least once during the first 4 years of life, and 55% (79) of them presented some type of DED. There was no statistically significant association between the variables analyzed. In conclusion, there was high prevalence of children with DED, and amoxicillin was the most commonly prescribed antibiotic.