A six-month-old Boston terrier presented with an extruded penis caudally, incompletely formed preputial sheath, bifid scrotum, retained testicle and deformity of the os penis. On physical examination, the urethral orifice was located on the surface of the perineum and a fibrous band was observed running from the grans to the urethral orifice on the perineum. The dog also had urethritis that was infected by ascending bacteria entering through the contaminated urethral orifice. Corrective surgery was undertaken to excise the external genitalia and retained testicle. The prepuce, penis and retained testicles were successfully excised. After the urethral orifice was cleaned periodically and antibiotics were administrated, recurrent urethritis disappeared. Using this therapeutic regime it is not necessary to reconstruct the anomaly located urethral orifice, if the location of urethral orifice is not the cause of recurrent urethritis and urinary incontinence.