A 28-year-old man was referred to our department for the management of recurrent hemospermia during the past 5 years. Genital examination and hormonal levels were normal. Semen analysis showed no change in volume and pH; however, hemospermia and asthenozoospermia were observed. Ultrasonography and computed tomography scan revealed the presence of a cystic lesion with calcification in the terminal part of seminal vesicles adjoining the prostate gland. The following vasography and endoscopic retrograde urethrography demonstrated 2 communicating cystic dilatations arising from the verumontanum. The diagnosis of cystic dilatation of the ejaculatory duct opening into an enlarged prostatic utricle was reached. Transurethral unroofing of the cyst was separately performed with a successful outcome. The characteristic of the 2 cystic dilatations was confirmed by pathologic examination. To the best of our knowledge, this is the first case of ectopic cystic dilatation of the ejaculatory duct opening into an enlarged prostatic utricle.