A 2-day-old preterm male neonate, a product of normal vaginal delivery, presented with respiratory distress, abdominal distension, and noticeable swelling of the penis along with dribbling of urine. On examination, the abdomen was distended with ascites, and a ballooned swelling of the anterior shaft of the penis was found. The neonate voided from normally site meatus and both testes were palpable in the scrotum (Fig. 1A). Renal function tests were deranged. Retrograde cystourethrogram showed scaphoid dilatation (like a balloon) of the ventral aspect of anterior and mid penile urethra confirming the diagnosis of congenital scaphoid megalourethra (Fig. 1B).

A congenital megalourethra is a rare abnormal enlargement of the male anterior urethra without distal obstruction because of dysgenesis of the penile corpus spongiosum with or without corpora cavernosa. ^[1] In 1989, Benacerraf et al. reported a case of congenital megalourethra diagnosed antenatally.^[2] In 1955, Nesbitt et al. defined megalourethra as “a congenital dilatation of penile urethra without distal obstruction. ^[3] Fewer than 100 cases have been published in the literature. ^[1] Dorairajan et al. described types of congenital megalourethra; scaphoid variant (associated with dysgenesis of the penile corpus spongiosum with bulging of ventral shaft of the urethra) and a fusiform variant (have deficient penile corpus spongiosum with corpora cavernosa with circumferential distension of anterior urethra).^[4]

There is penile functional obstruction caused by stasis of urine during micturition in the penile megalourethra due to a lack of penile supporting tissue without any anatomical obstruction of the urethra. ^[5] Megalourethra diagnosis can be made antenatally by sonographic features like cystic swelling in the perineal area of the fetus, fetal hydronephrosis, and bladder distended with a standard amount of amniotic fluid.