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Symptomatic mediastinal location of an extra-adrenal myelolipoma is extremely rare. We describe a 56-year-old female with unusual pain in the right lower posterior chest radiated to the neck and to the upper abdomen. Chest CT showed a lesion of 3.5 x 2.2 cm in the posterior mediastinum. Video-assisted resection was performed and the final pathologic examination revealed the presence of a myelolipoma. Herein we discuss the clinical presentation, the differential diagnosis and treatment of mediastinal myelolipoma.