Urethral coitus is a very rare sexual disorder seen in women either with intact or anomalous vagina such as vaginal agenesis or imperforate hymen. Only about 25 cases have been reported in the literature. We report a case of urethral coitus in a patient with surgically corrected ambiguous genitalia due to congenital adrenal hyperplasia. The patient had megalourethra and stenotic vaginal introitus. Megalourethra was corrected by plication technique. Correction of megalourethra with this technique has not been reported in literature up to now. To the best of our knowledge, the present case is the 26th of total cases with urethral coitus reported so far and the first case of urethral coitus associated with a stenotic vaginal introitus developing due to surgically corrected ambiguous genitalia.