Cystic dysplasia is a benign congenital lesion of the rete testis often associated with ipsilateral wolffian duct and ureteral abnormalities. We present a case of cystic dysplasia and associated anomalies in a 12-year-old boy. The workup revealed right testicular cystic dysplasia, ipsilateral renal agenesis, contralateral crossed renal ectopia, and vesicoureteral reflux. We performed right testicular-preserving cyst enucleation and ureteroneocystostomy. No testicular atrophy or new cyst formation was evident at 1 year of follow-up. This is the first reported case of cystic dysplasia associated with ipsilateral renal agenesis and contralateral crossed renal ectopia.
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