The occurrence of hypospadias in female patients is extremely rare. This paper concerns one woman with this anomaly, operatively treated at the Hospital of Hirosaki University. The case is a 22-year-old female who was admitted for left flank pain and frequent recurrence of urinary inflammation. Radical surgery, utilizing the skin flap of the anterior wall of the vagina and vestibulum for urethroplasty, was performed. The postoperative course was uneventful and the urinary infection was improved. This operative method is almost the same as the method employed for acquired urethrovaginal fistula. This case seems to belong to type 1 of female hypospadia according to Blum’s classification. The etiology of the congenital defect of the urethrovaginal septum still remains obscure. In regard to it, we would like to refer to a local developmental disturbance in type 1 from our clinical study of this patient.

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