Hypospadias is an anomaly of the male urogenital tract which occurs as a result of deficient closure of the urethral groove. Hypospadias is the most commonly seen urogenital anomaly in male neonates, but it is rarely diagnosed during prenatal period. Herein, a severe hypospadias case which was diagnosed by using 2D (two-dimensional) and 3D (three-dimensional) ultrasonography at 28 weeks of pregnancy with the aid of “tulip sign” finding was presented. 2D ultrasonography of 29 year old, primigravid woman at 28 weeks of gestation revealed an incurved, short penis with blunt tip located between the two scrotal folds and there was no additional anomaly. Findings were compatible with the “tulip sign” and case was diagnosed as isolated hypospadias. Isolated hypospadias was confirmed after delivery and surgical correction was planned. Prenatal diagnosis of hypospadias and relevant literature has been reviewed.