Cysts and canals found on the ventral surface of the penis have been ascribed to minor dysembryoplasia of the male genitalia. These lesions occur almost exclusively on the ventral surface of the penis along the median raphe and can extend from the urethral meatus to the anus. They are lined by stratified urothelium or squamous epithelium and they do not communicate with the urethra. Most cases are noted in early adulthood, they are usually asymptomatic but may become infected. Surgical removal is the adapted treatment. In this report we describe a case in which the urothelial origin of the cyst was documented by immunohistochemistry for cytokeratin 13.