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An unusual cause of female secondary infertility: Hypospadias. Female hypospadias is a very rare congenital anomaly and its impact on fertility has not yet been clearly defined. A 21-year-old woman with hypospadias was admitted with secondary infertility, dyspareunia, and urge symptoms. She was successfully treated with vaginal flap urethroplasty and broad spectrum antibiotics. Postoperatively, her symptoms resolved and she conceived spontaneously and aborted at her 17th gestational week following premature rupture of membranes suggesting infection. She then conceived spontaneously again and delivered a healthy term baby 30 months after the operation. Female hypospadias may cause chronic pelvic infections, urge symptoms, sexual dysfunction, hence infertility with time. After achieving normal anatomy by vaginal flap urethroplasty, treatment of chronic infections allows restoring normal urologic and sexual functions, and fertility.