CASE REPORT J Neurosurg 134:1271–1275, 2021 Neuromodulation of the lingual nerve: a novel technique Christopher E. Talbot, DO,1,2 Kevin Zhao, DO,1,2 Max Ward, BS,2 Aron Kandinov, MD,2,3 Antonios Mammis, MD,1,2 and Boris Paskhover, MD2,3 Departments of 1Neurological Surgery and 3Otolaryngology–Head & Neck Surgery, 2Rutgers New Jersey Medical School, Newark, New Jersey Acute injury of the trigeminal nerve or its branches can result in posttraumatic trigeminal neuropathy (PTTN). Affected patients suffer from chronic debilitating symptoms long after they have recovered from the inciting trauma. Symptoms vary but usually consist of paresthesia, allodynia, dysesthesia, hyperalgesia, or a combination of these symptoms. PTTN of the trigeminal nerve can result from a variety of traumas, including iatrogenic injury from various dental and maxillofa- cial procedures. Treatments include medications, pulsed radiofrequency modulation, and microsurgical repair. Although trigeminal nerve stimulation has been reported for trigeminal neuropathy, V3 implantation is often avoided because of an elevated migration risk secondary to mandibular motion, and lingual nerve implantation has not been documented. Here, the authors report on a patient who suffered from refractory PTTN despite multiple alternative treatments. He elected to undergo novel placement of a lingual nerve stimulator for neuromodulation therapy. To the best of the authors’ knowl- edge, this is the first documented case of lingual nerve stimulator implantation for lingual neuropathy, a technique for potentially reducing the risk of electrode migration. https://thejns.org/doi/abs/10.3171/2020.2.JNS193109 KEYWORDS neuromodulation; lingual; trigeminal; stimulation; burning mouth; face pain; surgical technique; peripheral nerve CUTE injury of the trigeminal nerve or its branches ing speech or eating.2 Thus, obtaining adequate symptom can result in posttraumatic trigeminal neuropathy relief in these patients is of the utmost importance in im- (PTTN), sometimes referred to as “trigeminal proving their quality of life. Although trigeminal nerve Anerve sensory neuropathy.” Affected patients suffer from stimulation has been reported for trigeminal neuropathy, chronic debilitating symptoms long after they have recov- V3 implantation is often avoided because of an elevated ered from the inciting trauma. Symptoms vary but usually migration risk secondary to mandibular motion, and lin- consist of paresthesia, allodynia, dysesthesia, hyperalge- gual nerve implantation has not been documented.9,10 To sia, or a combination of these clinical symptoms.1,2 the best of our knowledge, we present the first documented PTTN of the trigeminal nerve can result from a variety case of lingual nerve stimulator implantation for lingual of traumas, including iatrogenic injury from various dental neuropathy, and a technique for potentially reducing the and maxillofacial procedures. Lingual pain and paresthe- risk of electrode migration. This novel procedure required sia have also been reported in patients with traumatic basi- a multidisciplinary approach between otolaryngology (ear, lar skull fracture involving the foramen ovale.3 Surgery of nose, and throat [ENT] surgery) and neurosurgery for their the mandibular third molar is the most common iatrogenic respective familiarity with transoral surgery and neuro- injury, with a reported frequency ranging from 0.5% to modulation. 0.6%.1,4,5 Sagittal split mandibular ramus osteotomy, im- plant surgery, endodontic treatment, and various oral pro- Case Report cedures can also injure the lingual nerve.1,4 Lingual and/or inferior alveolar nerve injury is a known History and Physical Examination risk of many oral and maxillofacial surgeries, and while A 58-year-old man was referred to the functional neuro- the vast majority (> 90%) of cases are temporary, those surgery and otolaryngology clinic for tongue pain, numb- lasting more than 6 months are deemed permanent.6–8 The ness, and dysesthesia refractory to medical management. severity of symptoms can vary; however, up to 70% of He reported a prolonged course of pain in his tongue and patients with PTTN may report pain significantly impair- along the left anterior floor of the mouth up to the lingual ABBREVIATIONS ENT = ear, nose, and throat; PTTN = posttraumatic trigeminal neuropathy. SUBMITTED November 17, 2019. ACCEPTED February 24, 2020. INCLUDE WHEN CITING Published online April 24, 2020; DOI: 10.3171/2020.2.JNS193109. ©AANS 2021, except where prohibited by US copyright law J Neurosurg Volume 134 • April 2021 1271 Unauthenticated | Downloaded 10/07/21 09:00 AM UTC Talbot et al. FIG. 1. Illustrations showing electrode placement from medial (A) and posterior (B) perspectives with labeling of anatomically significant structures. Copyright Boris Paskhover. Published with permission. alveolar ridge. The pain was exacerbated by talking and assistance in appropriate lead placement via a submental stress but decreased with mastication. These symptoms approach (Video 1). began after genioglossus advancement with lingual fren- VIDEO 1. Intraoperative video clip of a transoral exposure using an ulum release 7 years earlier. At presentation, the patient endoscope demonstrating exposure of the left lingual nerve before had already been evaluated by multiple neurosurgeons, and after placement of a stimulator electrode. Copyright Rutgers- interventional pain specialists, otolaryngologists, oral sur- NJMS, Newark, NJ. Published with permission. Click here to view. geons, and dental practitioners without significant symp- A 2-cm incision was made in the mucosa along the medial tom relief. He had undergone pulsed radiofrequency abla- aspect of the left mandibular ramus. With the aid of a rig- tion along the left ascending ramus, which improved his id endoscope to improve visualization, careful soft-tissue symptoms transiently for 2 days. He had received multiple dissection was continued toward bone. The periosteum local anesthetic injections targeting the lingual nerve with was vertically incised, and the ascending ramus was fol- only temporary relief. He was not a candidate for nerve lowed rostrally in a subperiosteal plane until the inferior resection and reconstruction since he was out of the opti- alveolar nerve was identified entering the inferior alveolar mal time frame.11 canal. This nerve was traced proximally until its intersec- He subsequently underwent transoral partial ablation of tion with the lingual nerve was identified. The lingual the left lingual nerve, which resulted in mildly increased nerve was carefully isolated from surrounding soft tissue, numbness and transient incisional pain but no decrease and scar tissue near the nerve, from prior ablations, was in neuropathic pain. The symptoms from this mild ab- removed. The nerve’s course was traced along the floor lation dissipated over the following 2 months. Seeking of the mouth to confirm continuity and integrity, and no more aggressive treatment, the patient underwent repeat neuromas or lesions were identified along this path. The radiofrequency ablation without significant relief but with dissection was then carried 2–3 cm rostral to the inferior increased numbness. For persistent symptoms and severe alveolar branch point, toward the foramen ovale, to iden- discomfort refractory to maximal noninvasive and inva- tify a portion of the nerve not previously manipulated, for sive therapies, the patient was deemed a candidate for a optimal electrode contact. novel approach to nerve stimulation, which was performed A 1.5-cm submental incision along a natural skin crease in two stages. The patient had not undergone any previous was made, and the dissection was continued along the in- attempts at trigeminal nerve or cervical spinal cord neu- ferior edge of the mandible in a subperiosteal plane toward romodulation. the left floor of the mouth. The dissection was advanced Gross examination of the tongue and oral cavity was below the mylohyoid musculature so that its attachments unremarkable. Spasms of the intrinsic tongue muscles would not be disrupted. A tunneling sheath was then placed were not reported by the patient or observed. There were with endoscopic assistance, and a linear-array percutane- no motor deficits of the tongue or muscles of mastication. ous lead (Medtronic) was introduced into the dissected Sensory examination was significant for limited numb- pocket (Fig. 1). The lead was pushed into place while pal- ness over the left anterolateral margin of the tongue and pating its path along the floor of the mouth. The stimulator accompanied by a surrounding region of dysesthesia. was situated posterior and parallel to the vertical segment of the lingual nerve, and electrodes were confirmed to be Operative Technique in contact with the nerve under endoscopic visualization To begin, the ENT surgeon performed a careful trans- (Fig. 2). Contacts of the stimulator were confirmed cephal- oral dissection to the lingual nerve for identification and ically with contacts along the main branch of V3. 1272 J Neurosurg Volume 134 • April 2021 Unauthenticated | Downloaded 10/07/21 09:00 AM UTC Talbot et al. FIG. 2. Endoscopic visualization of the stimulator electrode alongside the left lingual nerve. The left medial mandibular cortex is marked with an asterisk. The electrode was then anchored to the submental peri- osteum to permit the electrode to move as a single unit with the mandible in an effort to minimize the risk of elec- trode migration. Electrode wires were then