Introduction: Schwannomas are benign neurogenic tumors arising from neural sheaths and rarely develop in the sublingual space. We report the case of an ancient sublingual schwannoma in an asymptomatic 12-year-old patient. Furthermore, we propose a review of the literature on non-vestibular schwannomas of the sublingual region.Materials and Results: We performed a complete excision of the lesion through a submandibular transcervical approach, sparing the left sublingual and submandibular glands, lingual nerve, and hypoglossal nerve. There was no need to perform a sural nerve graft to restore nerve function. The nerve of origin was identified intraoperatively as the mylohyoid nerve and was sacrificed during the en-block removal of the lesion. As of today, there are still multiple issues in the diagnosis and management of head and neck non-vestibular schwannomas. Preoperative evaluation through fine-needle aspiration cytology has an accuracy of only 20%. Identification of the nerve of origin is complex but critical to preserve nerve function after treatment.Conclusion: An accurate preoperative evaluation allows to make an informed decision whether to undergo surveillance or surgery, with potential nerve grafting after removal of the lesion. Definitive diagnosis is often possible only after surgery, with clinical observation and histopathological examination of the surgical specimen.