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Sclerosing perineurioma is an extraneural soft tissue lesion that has been reported in the extremities of young adults. It is composed of oval epithelioid or plump spindled cells scattered to form ill-defined fascicles in a storiform, whorled, or trabecular pattern and dermal sclerosis represented by thickened collagen bundles in a lamellar array that express epithelial membrane antigen and are negative for S-100 protein. The treatment is surgical excision, and no recurrence or metastases are expected. Herein is presented the first case of intraoral sclerosing perineurioma of the lower lip, emphasizing its histopathologic and immunohistochemical features.