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We present a rare case of an intramedullary osteolipoma without any extramedullary connections or coexisting spinal dysraphism. A 48-year-old woman presented with back pain and pain around the lateral aspect of the left thigh. Magnetic resonance imaging (MRI) revealed a heterogeneous hyperintense and hypointense region on T1 and T2-weighted imaging. A cavernous hemangioma was suspected, and the patient was admitted for surgical resection. The histological diagnosis was an intramedullary osteolipoma.MRI with fat suppression and computed tomography are recommended for the diagnosis of an intramedullary osteolipoma, but a definitive diagnosis requires histological examination.