Megalourethra is a rare congenital malformation characterized by dilatation of the penile urethra. We review 6 previously unreported cases and 35 cases reported in the literature. The distinction between scaphoid and fusiform megalourethra seems arbitrary, and the disorder is better viewed as a spectrum rather than as 2 distinct entities. Significant concomitant upper urinary tract abnormalities may be present in any patient with megalourethra, a fact that should lead to the routine investigation of the upper urinary tract in all patients with this developmental anomaly.