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Duplication of the ureter is a common congenital malformation, which unfortunately may be accompanied by unpleasant and challenging pathologies. Hereby, a rare case of a patient with obstructive urolithiasis secondary to undiagnosed complete ureteral duplication is presented. A single large calculus in the vesicoureteral junction was obstructing both duplicated ureters. The aim of this article was to discuss both the challenges arising from this clinical entity and the diagnostic approaches. In such complicated cases combined with suspected pyelonephritis or severe hydronephrosis, the option of an urgent lithotripsy should be considered. The obstructed orifices are often inflammatory, hindering their stenting. Asymptomatic and undiagnosed patients with completely duplicated ureters are prone to severe complications. Thus, early screening of these patients is an imperative need for the clinician.

Incidental finding of unilateral complete duplication of ureter in a patient with large ureteric calculus obstructing both left limbs