Congenital prepubic sinus is a rare anomaly: only five pediatric cases
have to our knowledge been reported to date. We report two further ca
ses with the sinus extending from the overlying skin through the rectu
s fascia to the anterior bladder wall. In our first case the opening o
f the sinus was just 3 cm below the midline inferior to the navel, wit
h concentric, thin smooth-muscle layers. This lesion may represent a m
ild forme fruste of a midline closure defect. In our second case, the
sinus was lined by transitional epithelium in its distal portion with
stratified squamous epithelium near the surface, and was also surround
ed by a thin smooth-muscle layer. These two cases may represent a vari
ant of epispadiac urethral duplication. Our observations suggest that
congenital pre-pubic sinus and urethral duplication are related embryo
logically.