Urethral duplication and megalourethra are very rare anomalies and their concomitant presence is extremely rare, with only a few published cases. We present a complex case of complete urethral duplication with dorsal megalourethra that was severely stenotic in its bulbar part and meatus, with the ventral urethra atretic distally and dilated proximally. Both the corpus spongiosum and the cavernosum were missing. He had associated upper urinary tract abnormalities. Urethral patency was restored successfully by meatoplasty, staged buccal mucosa graft urethroplasty, and tailoring of the megalourethra. This report is unique regarding the use of a buccal mucosa graft for urethral reconstruction in patients with associated urethral duplication and megalourethra.