To accommodate the small size of the infant urethra, finer, more flexible tubes are often used for urinary catheterization in the pediatric intensive care units. These tubes have the ability to knot in the bladder, occasionally requiring surgical removal. The mechanism of knotting appears to result from excessive intravesical catheter coiling, and as the bladder decompresses the catheter tip can migrate through a coil thereby creating a knot. Review of the literature from 1975 to 2000 identified 19 cases of urethral catheter knotting in the pediatric bladder with two reports of prostatic urethral involvement. Herein, we describe the first reported instance of catheter knotting within the penile urethra and describe the surgical technique employed for its removal.