To our knowledge we report the first case of retroperitoneal angiomyolipoma located at the left ureterovesical junction associated with ipsilateral renal agenesis. Radiologically, the mass had the characteristics of a cystic soft tissue tumor. Final diagnosis was determined by histopathological examination, which revealed the specific characteristics of angiomyolipoma. The tumor is theorized to be a result of developmental malformation of fetal mesenchymal elements along the ureteral bud during fetal development since it was associated with ipsilateral renal agenesis.
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