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The purpose of the study was (1) to investigate gender role behaviors of boys with hypospadias compared with groups of unaffected boys and girls using parental reports and direct observations; and (2) to directly observe effects of socialization (mothers’ presence) on children’s gender role behaviors. Ages of 19 children with hypospadias ranged from 3 to 7 years, and each of them were matched to controls of unaffected boys and girls by age. All the children participated with their mothers. Children’s gender role behaviors and their mothers’ behaviors were evaluated using an observation coding system. Mothers also completed questionnaires regarding their children’s gender role behaviors. Results indicated no atypical gender role behavior for the boys with hypospadias and no direct effects of socialization on their gender role behaviors. However, differences were found in negative communicative behaviors between boys with hypospadias and unaffected boys, suggesting a possible role of socialization.