ABSTRACT
BACKGROUND: Urethral duplication and megalourethra are rare urethral anomalies. However, the concomitance of urethral duplication and double megalourethra has not been reported previously. CASE REPORT: A newborn was presented with penile swelling during voiding. Physical examination revealed a retractable foreskin and two external meatus of a double urethra. Retrograde urethrography demonstrated two complete megalourethras. Urethro-urethrostomy and urethroplasty were performed when the patient was 10 months old. The patient was followed up for one year without any urinary problems and has good cosmetics and urinary continence. CONCLUSION: The concomitance of these two rare anomalies and more importantly its surgical treatment makes this case report unique and valuable.