Objective: To report the clinical outcome of urethral reconstruction by cultured urothelial or oral mucosa cells for tissue-engineered urethroplasty. Methods: We systematically searched for studies reporting the use of tissue-engineered techniques for hypospadias and urethral stricture repair in humans in PubMed and Embase (OvidSP) through January, 1990 to June, 2018. We excluded studies based on titles that clearly were not related to the subject, studies in which tissue-engineered biomaterial were used only in laboratory or experimental animals, and in the absence of autologous cultured epithelial cells. Studies were also excluded if they were not published in English, had no disease background and adequate follow-up. Finally, we search all relevant abstract presented at two of the main urological meetings in the last 10 years: European Association of Urology (EAU) and American Urological Association (AUA). Results: A total of six articles, reporting the clinical use of tissue-engineered techniques in humans, were fully reviewed in our review. The epithelial cells were harvested from the urethra (10 patients), the bladder (11 patients) and the mouth (104 patients). The tissue-engineered grafts were used in children for primary hypospadias repair in 16 cases, and in adults for posterior and anterior urethral strictures repair in 109 cases. Tissue-engineered grafts were showed working better in children for primary hypospadias repair than in adults for urethral strictures repair. Conclusion: One hundred and twenty-five patients received tissue-engineered urethroplasty using cultured epithelial cells for primary hypospadias or urethral strictures repair. The studies demonstrate a high degree of heterogeneity respect to epithelial cells (from urethra, bladder, and mouth), type of scaffold, etiology, site of urethral stricture, number of patients, follow-up and outcomes.