Congenital prepubic sinus with dorsal penile curvature: a case report and literature review

Background: Congenital prepubic sinus (CPS) is a rare congenital anomaly and widely thought to be a variant of urethral duplication. Histological examination of this case gives a clue to this theory. CPS with dorsal penile curvature has been reported in previous publications, but their procedures to correct the curvature are different from this case.

Case presentation: A 10-year-old boy complained of the pain in the dorsal base of the penis. Physical examination revealed an accessory meatus located in the midline of the dorsal proximal penis and moderate dorsal penile curvature with deficient dorsal foreskin. Imaging examination showed that the meatus did not communicate with either normal urethra or urinary bladder, and ended blindly at the level of the symphysis pubis. The intact 4-cm-long sinus was completely separated and excised. Penile curvature was corrected after the dorsal proximal fibrous cord was detached. Histological examination confirmed the diagnosis of urethral duplication.

Conclusions: The histological result of this case supports the theory that CPS is a variant of the dorsal urethra. Moreover, this case indicates that the curvature in patients with CPS may be caused by the dorsal fibrous cord at the beginning and the operation should be conducted at an early age to avoid further development of the curvature during puberty.