Rectourethral fistula after anoplasty is a common disease. This study reports on a 7-year-old male child with rectourethral fistula after anoplasty. The main clinical manifestation was leakage of urine from the anus. The child had undergone the anoplasty in other hospitals due to no anus. Routine voiding cystourethrography showed urethral fistula. The rectourethral fistula neoplasty with transperineal approach was performed with a surgical duration of 64 minutes. During surgery, there were no obvious complications such as wound infection, wound dehiscence, rectal retraction, etc. The fistula was completely healed 2 weeks after the surgery. On the 2nd month after surgery, the appearance of anus was satisfactory, with no fecal incontinence. The child was followed up for 2 years, and had normal urination. The surgical operation is simple and feasible and the outcome was satisfactory.