Cystic dysplasia of the rete testis (the network of straight seminiferous tubules within the mediastinum testis leading to the efferent ductules) is a rare congenital anomaly. There are only 15 cases reported in the literature to date and only one report of the US findings [1-5]. This report highlights the characteristic appearance of the lesion on ultrasound examination and its association with renal abnormalities in two patients.