The authors report on two patients who had fusiform megalourethra with unusual features. The first patient had left hydronephrosis with duplication of the ureter, oblique sagittal septum in the urinary bladder, and Y duplication of urethra with megalourethra of both channels. The second had features of the abdominal muscle deficiency syndrome, with anterior ectopic anus, rectourethral fistula, and fusiform megalourethra. Both patients were treated by preliminary urinary diversion. The first patient subsequently had staged reconstruction. The other patient died of urinary sepsis. The authors believe that preliminary urinary diversion followed by planned staged reconstruction offers hope for the reconstruction and rehabilitation of these cases.