Transitional cell carcinoma of the prostatic urethra was first described and reported in 1963. Primary urothelial carcinoma of the prostate is an extremely rare disease, often misdiagnosed with other entities, particularly with urothelial carcinoma of the bladder involving the prostatic tissue. There are only dozens of cases of primary urothelial carcinoma in the prostate reported in the literature. The prognosis after diagnosis is reported to be poor and there is no consensus among experts concerning its proper treatment and follow-up. Herein, we report two cases of patients diagnosed with this entity with a median follow-up of 14 months.