Introduction Myelolipomas are uncommon, benign tumors composed of mature adipose tissue and hematopoietic elements. They mostly occur in the adrenal glands, but extra-adrenal myelolipomas have also been reported in other locations such as the presacral region, retroperitoneum, pelvis and mediastinum. Here, we present a case of an extra-adrenal myelolipoma in a rare site: the renal parenchyma. To the best of our knowledge, it is only the third case reported in this unusual location. Case presentation We report a case of primary myelolipoma occurring in the kidney of a 55-year-old Moroccan man. We describe the radiological and clinicopathologic features of this unusual tumor with a review of the literature, and we discuss differential diagnosis of retroperitoneal myelolipomas. Conclusion This case is noteworthy because the tumor site was unusual. Although renal myelolipoma is rare, it should be considered in the differential diagnosis of lesions in this site.
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