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Severe penile torsion of 180° associated with hypospadias is a rare entity. Knowledge of penile anatomy and pathology are necessary as the diagnosis could be missed.    Case presentation  We report a case of severe 180° penile torsion with distal hypospadias that was mistaken for an epispadias which was corrected with surgery.    Conclusion  Tubularized incised plate urethroplasty and dartos flap rotation provided satisfactory result for this association.

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180° congenital penile torsion with distal hypospadias mistaken for an epispadias: Optimal outcome with tubularized incised plate urethroplasty and dartos flap rotation