Congenital megalourethra is a rare urogenital malformation that is usually associated with lower urinary tract obstruction. In association with hypoplasia or deficiency of the corpus spongiosa and/or corpus cavernosum, a cystic dilatation of penile urethra is generally observed. We present a case of congenital megalourethra diagnosed by fetoscopy and ultrasonography at 17 weeks of gestation. Fetoscopic investigation demonstrated a balloon dilatation of the penis with a complete obstruction of the urethral meatus, which could distinguish a fusiform type congenital megalourethra from other disorders. Autopsy subsequent to the termination of pregnancy revealed a complete deficit of both corpus spongiosa and corpus cavernosum.
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