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The congenital prepubic sinus is a rare type of fistula with a broad range of anatomical and histological diversity. We report the case of an 8-month-old boy with a congenital prepubic sinus where immunohistochemistry studies showed only squamous epithelium and no transitional epithelium. The etiology of this disorder is still unclear and the most recent case reports suggest that the fistula might be a variation of a dorsal urethral duplication. We recommend performing immunohistochemical studies to further clarify the etiology, as the fistula may be caused by different developmental disorders.

El seno prepúbico congénito es un tipo de duplicación uretral poco frecuente, de etiología desconocida. Se han propuesto distintas teorías embriológicas, como así también diversas clasificaciones. La mayoría de los casos se diagnostican en la infancia, la cistouretrografía retrógrada y miccional es el estudio fundamental, tanto para confirmar el diagnóstico como para establecer el tipo de anomalía y la mejor opción terapéutica. Presentamos un caso de seno prepúbico congénito en un varón de 39 años de edad, diagnosticado por secreción purulenta a nivel de un meato accesorio en base del pene. The prepubic congenital sinus is a rare type of urethral duplication of unknown origin. Different embryological theories and classifications has been proposed. Most cases are diagnosed during childhood. The Retrograde Urethrocystography is very important as it determines the diagnosis and helps choosing the best treatment option. We present a case of a prepubic congenital sinus in a 39 years old male who presented with purulent discharge from an accessory meatus in the base of the penis.