Background: Diphallia is a very rare anomaly and seen once in every 5.5 million live births. True diphallia with normal penile structures is extremely rare. Surgical management for patients with complete penile duplication without any penile or urethral pathology is challenging.
Case Report: A 4-year-old boy presented with diphallia. Initial physical examination revealed first physical examination revealed complete penile duplication, urine flow from both penises, meconium flow from right urethra, and anal atresia. Further evaluations showed double colon and rectum, double bladder, and large recto-vesical fistula. Two cavernous bodies and one spongious body were detected in each penile body. Surgical treatment plan consisted of right total penectomy and end-to-side urethra-urethrostomy. No postoperative complications and no voiding dysfunction were detected during the 18 months follow-up.
Conclusion: Penile duplication is a rare anomaly, which presents differently in each patient. Because of this, the treatment should be individualized and end-to-side urethra-urethrostomy may be an alternative to removing posterior urethra. This approach eliminates the risk of damaging prostate gland and sphincter.