Primary adrenal leiomyosarcomas (LMS) are extremely rare mesenchymal tumors. To the best of our knowledge only 12 cases (9 cases of conventional and 2 cases of pleomorphic LMS) have been reported in the literature until the present date. Herein we report a case of pleomorphic LMS in a 47-year-old woman who presented with severe abdominal pain. Computed tomography showed a large heterogeneous left adrenal mass abutting the renal vessels. The adrenal mass was resected and histopathologic examination revealed a highly pleomorphic malignant spindle cell neoplasm diffusely infiltrating the adrenal parenchyma with an immunoprofile consistent with that of a leiomyosarcoma. Clinical management and subsequent follow-up of this patient is presented.
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