Congenital prepubic sinus is a tract originating in the skin overlying the base of the penis. Its embryologic basis is still debated. We present a 9-month-old boy with a recurrent muco-purulent discharge from a tiny opening in the midline prepubic area on the base of the penis. Examination revealed a ventrally hooded prepuce, dorsal chordee, penile torsion to the left, and a 3-mm-diameter prepubic sinus 1 cm from the base of the penis. Histology of the excised tract showed a transitional epithelium. We believe our case is an epispadiac variant of dorsal urethral duplication.
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