Perineurioma is a rare benign nerve sheath tumor derived of perineurial cells. There are two main types of perineurioma which differ histologically and clinically: intraneural and extraneural (soft tissue) perineurioma. Intraoral perineurioma is extremely rare. This report describes a case of multiple orofacial intraneural perineuriomas in a patient with hemifacial hyperplasia and discusses clinical, histologic, and ultrastructural features as well as differential diagnosis of perineuriomas, with emphasis on intraneural perineurioma.