To report a case of cystic dilatation of the prostatic utricle.A case of cystic dilatation of the prostatic utricle associated with right renal agenesis in a 26-year-old patient who consulted for recurrent orchiepididymitis and a history of hemospermia is presented.Physical examination showed a hypogastric tumor mimicking a vesical globus that was palpable on digital rectal examination. Ultrasonography and urethrocystoscopy were decisive in the diagnosis of this anomaly. The utricle was removed by the transperitoneal approach. Gross examination showed opening of both deferent ducts and right ureter (blind-ending) into the utricle.The pathological characteristics of this anomaly permitted its identification as cystic dilatation of the prostatic utricle, a condition that should be taken into account in the differential diagnosis of pelvic tumors in the male.