We report on a symptomatic anterior intraurethral prostatic cyst in a 46-year-old man without clinical evidence of benign prostatic hyperplasia. The anterior location of this cyst makes it unique to all previously reported cases of prostatic cysts which are located posteriorly. Transurethral resection of the cyst with limited resection of the anterior prostatic tissue at the base of the cyst was performed with successful resolution of voiding symptoms. In the absence of lateral lobe hypertrophy, standard transurethral resection of the prostate should be avoided to ensure preservation of erectile and ejaculatory function.