The surgical treatment of severe hypospadias can be very challenging. In selected cases with severe lack of tissue, cultured autologous urothelial cells can be used to create a transplanted neourethra. The algorithm for using tissue-engineering techniques for the surgical repair of the male urethra includes 3 main steps. First, the autologous urothelial cells are harvested from the patient, e.g. by bladder washing through a catheter with sterile saline. Second, the cells are propagated and expanded in the laboratory environment. Finally, the cells are transplanted back to the patient in the final surgical repair. We have used this technique in a clinical setting in severe hypospadias. Transplants for creation of the neourethra were used ventrally in an on-lay fashion with remaining skin dorsally in the urethra. Our follow-up shows a good clinical result including data concerning voiding position, urinary flow, artificial erection, cosmetic appearance, urethroscopy and biopsies. Tissue-engineering techniques can, however, benefit from further improvement and, thus, be developed for additional applications. We conclude that in hypospadias repair with cultured autologous urothelial cells is an option for the surgical treatment when there is a lack of local tissue for the repair.