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Single-system ureterocele is an important, although uncommon cause of hydronephrosis and renal dysplasia in infants and children. Single-system ureterocele is distinguished clinically from the more common duplex-system ureterocele by its frequent occurrence in boys and its association with multicystic dysplastic kidney. Because these ureteroceles are frequently small and have a propensity to evert at VCUG, they can be mistaken for paraureteral diverticula.