OBJECTIVES: The present case is a diffuse isolated plexiform neurofibroma of the tongue that was not associated with neurofibromatosis that we treated with intraoral surgery. STUDY DESIGN: A case report. METHODS: We present a 5-year-old girl with isolated plexiform neurofibroma of the tongue with infiltration of the tongue base and review treatment approaches for this very rare tumor. RESULTS: An intraoral approach appears to be a good alternative method that allows both an acceptable exposure for total excision and limited postoperative sequelae. CONCLUSIONS: Isolated plexiform neurofibroma of the tongue is very rare. It causes a massive macroglossia that is progressive with advance of years and disturbs the patient's speech, occlusion, and tongue movements. Involvement of the tongue may be associated with infiltration into the deep structures of the neck by the tumor, thereby presenting considerable operative difficulties.
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