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Angiomyolipoma (AML) consists of <10% of all renal tumors and is the most common benign mesenchymal neoplasm of the kidney. It arises from the renal cortex and extends toward perirenal fat. Fat-poor AML as well as those arising from rare locations may pose diagnostic difficulties. In our case, we report a rare presentation of this tumor arising from the renal sinus extending toward the pelvis thereby leading to an alternative diagnosis of transitional cell carcinoma of renal pelvis.