In this genetic study of atypical gender role development, parents of 5,799 twin pairs, ages 3 and 4, rated their twin children's masculinity and femininity. Boys were selected as gender atypical if they were highly feminine (top 5%, 10%, or 15%) relative to other boys, and girls were selected if they were highly masculine relative to other girls. Gender-atypical boys and girls were each divided into 2 groups: fully gender atypical (e.g., feminine boys also low on masculinity) and partially gender atypical (e.g., feminine boys who are not low on masculinity). DeFries-Fulker (DF; J. C. DeFries & D. W. Fulker, 1985, 1988) extremes analysis yielded moderate group heritability and substantial shared environment effects for atypical gender role behavior. However, for fully gender-atypical girls, group heritability accounted for most of the variance, and shared environment had no effect. The results are discussed in light of past studies and potential implications for atypical gender development.