Urethral duplication is one of the rarest congenital urinary tract anomalies, commonly seen in males and there has been fewer than 200 cases reported until now. We propose that the nomenclature that has been used for these anomalies may be changed as urethral misalignments. Although, the etiology is not clear, and the exact embryology of the malformation has yet to be defined; it seems that the anomaly is not a duplication of a tissue but in fact a problem which prevents the penile and glanular urethra meet with each other. Therefore, misalignment of the urethra makes more sense than urethral duplication in describing these anomalies. We reported four patients with urethral misalignment and their preferred surgical treatments. One of the cases was a collateral urethral duplication, and there are only nine other cases that have been reported in the literature. Urethral duplication or as we suggest, urethral misalignment is a rare anomaly without a complex treatment approach providing that the exact anatomy of the both urethras and their relation with each other are well understood by the surgeon. Once this has been satisfied the necessary surgical procedures are usually simple and easy to perform for many plastic surgeons.

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