Osteolytic lesions are highly suggestive of metastatic disease or in rare instances may result from primary extradural meningiomas. Herein, we describe a rare case of a patient who presented with a large area of osteolysis on the right orbital roof. An excisional biopsy revealed that the lesion was associated with the presence of hyperplastic intra-osseous meningothelial tissue. To our best knowledge, this is the first evidence that ectopic nonneoplastic meningothelial cells may induce osteolysis on the orbital walls.
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