Objective: Congenital anomalies or malformations of the urethra that involve number are extremely rare and only 7 cases of trifurcation of the urethra and under 500 cases of urethral duplication have been described.
Methods: In the case we present, urethral duplication was diagnosed in adulthood, even though glans malformation with 2 urethral orifices was clearly evident and only 1 was functional.
Results: The condition is usually diagnosed in childhood due to the presence of 2 urethral meatus or from double stream if both are functional; at other times, the condition is diagnosed from complications that trigger infections or obstruction or if it is associated with more extensive malformations.
Conclusions: Urethral duplication is an extremely rare birth defect, and diagnosis in adulthood is even more uncommon. No single embryologic argument explains all the anatomic variants of urethral duplication. The treatment of the urethral duplication should be individualized according to the type of duplication and the clinical symtoms.