Objectives.: Fibrous hamartoma of infancy (FHI) is an uncommon subcutaneous proliferative lesion usually found in the upper portions of the body, particularly in males during the first 2 years of life. We report 2 cases of FHI in the genital region. Methods.: We retrospectively reviewed the charts of children treated at Children's Hospital and Health Center, with FHI located in the genital region. Results.: We report 2 cases of FHI in the genital region, 1 involving the scrotum and 1 the labium majus. Conclusions.: FHI should be considered in the differential diagnosis of genital masses in children. The histologic features are characteristic, local excision of the tumor is curative, and the local recurrence rate is low.