Persistent urogenital sinus (UGS) is a developmental anomaly. It represents one of the most complex problems that a pediatric surgeon may deal with. We report the case of a patient with UGS treated at 3 years of age by anterior sagittal transrectal approach and en bloc sinus mobilization. The procedure was performed with the patient prone with the initial idea of performing an anterior sagittal transrectal approach. The described approach allows an excellent anatomical view with a midline muscle sparing incision, along with an easy identification of the vaginal confluence with the benefit of avoiding dissection between the urethra and vagina.