Delayed Hypoglossal Nerve Palsy After Halo-Vest Immobilization in a Patient With C1-C2 Tuberculosis: A Case Report.

CASE: A 48-year-old man presented with neck pain and torticollis without any neurodeficit. He was diagnosed with C1-C2 tuberculosis with left C1-C2 joint destruction and was immobilized with a halo vest and started on antitubercular treatment (ATT). At the 3-month follow-up, he presented with left hypoglossal nerve palsy (HNP). He responded to ATT with complete healing of C1-C2 lesion and good correction of deformity but persistent left-sided tongue deviation at the 2-year follow-up. CONCLUSION: HNP may occur as a complication of conservative management of craniovertebral junction tuberculosis (CVJ TB). Careful neurological assessment and monitoring must be performed while correcting deformities in CVJ TB using halo-vest immobilization.

Idiopathic Isolated Unilateral Hypoglossal Nerve Palsy: A Report of 2 Cases and Review of the Literature.

Hypoglossal nerve palsy (HNP) is a common finding in neurologic diseases when associated with other cranial nerve palsies or further pathology and exhibits characteristic clinical manifestations, including unilateral atrophy of the musculature of the tongue. It occasionally appears as the initial or solitary sign of an intracranial or extracranial space-occupying lesion, head or neck injury, or vascular abnormality of the internal carotid artery. There are few cases of idiopathic isolated unilateral HNP, which should be diagnosed through exclusion. This report describes 2 patients who had different outcomes and presents a literature review of idiopathic isolated unilateral HNP. Case 1 was a 71-year-old man who was referred with a 1-month history of dysphagia and speech impairment. Intraoral examination disclosed marked left-side hemiatrophy of the tongue and deviation toward the left on protrusion. At coronal Tl-weighted magnetic resonance imaging, left-side hemiatrophy of the tongue was clearly visible through deviation of the median septum to the left. The patient was diagnosed with idiopathic isolated unilateral HNP through exclusion and was treated with steroids and mecobalamin, but he did not recover. Case 2 was a 32-year-old man complaining of tongue weakness for 2 days. On examination, left HNP was evident, with deviation of the tongue to the left on protrusion. He was diagnosed with idiopathic isolated unilateral HNP through exclusion and was treated with steroids. After 3 weeks, the patient had completely recovered. To the best of the authors' knowledge, this is the first detailed literature review on idiopathic isolated unilateral HNP. This condition is very rare but should be considered for diagnosis. It warrants a thorough and stepwise approach for etiologic diagnosis.

Malignant peripheral nerve sheath tumor of the tongue with an unusual pattern of recurrence.

Malignant peripheral nerve sheath tumor (MPNST) of oral cavity is an extremely uncommon malignancy. Less than 15 cases have been reported since 1973 though none of them describes a distant metastasis. We present a rare case of MPNST of the tongue who presented with features of hypoglossal nerve palsy. Incisional biopsy showed a malignant spindle cell tumor in the sub-epithelial connective tissue. The tumor cells were immune-positive for S-100. He underwent surgery followed by adjuvant chemo-radiation. Later the disease recurred in the form of isolated pelvic bone metastasis. Palliative chemotherapy was offered to him. With this case report we intend to refer to such unusual presentation and pattern of recurrence in a MPNST of tongue.

Isolated tubercular hypoglossal nerve paralysis.

Hypoglossal nerve palsy is not an uncommon neurological finding but primary nasopharyngeal tuberculosis (TB) presenting as hypoglossal nerve palsy is very rare. A 31-year-old woman presented with headache and progressive tongue deviation towards the right side. Diagnostic nasal endoscopy revealed soft tissue mass lesion on the posterior wall of nasopharynx while MRI revealed isointense tumour in nasopharynx with normal hypoglossal nerve and brain. Histopathological examination found TB. We discuss the clinical challenges and possible pathogenesis of this rare clinical entity.

High-dose intravenous steroid regimen for radiation-induced hypoglossal nerve palsy.

BACKGROUND: Hypoglossal nerve palsies are infrequent complications of head and neck radiotherapy. Treatments focus on maintaining function and prevention of abnormal airway-related swallowing events. METHODS: A patient with longstanding cranial neuropathies, including bilateral hypoglossal involvement, secondary to chemoradiotherapy for nasopharyngeal carcinoma, experienced repeated episodes of life-threatening complications. Initially, 2 courses of 2 weekly 24-hour intravenous methylprednisolone (IVMP) infusions were administered 2 years apart. We report the results of a third course comprising 5 weekly cycles. RESULTS: Patient-reported outcomes revealed significant improvement in swallowing function, speech, and psychosocial status. Airway invasion during swallowing and pharyngeal retention were assessed videofluoroscopically and evaluated using the Penetration-Aspiration Scale (PAS) and a residue rating scale, respectively. PAS ratings after infusions 2 and 5, improved dramatically from baseline and were maintained at 1-year follow-up. CONCLUSION: High doses of IVMP may improve radiation-induced neuropathies. Further testing in similar patients is needed to prove reproducibility. © 2016 Wiley Periodicals, Inc. Head Neck 39: E23-E28, 2017.

Asymmetric Acute Motor Axonal Neuropathy With Unilateral Tongue Swelling Mimicking Stroke.

A 60-year-old man presented with acute onset of left hemiparesis and left hypoglossal nerve palsy with ipsilateral tongue swelling. He then progressed to tetraparesis in a few days. Cerebrospinal fluid showed cell protein dissociation. A nerve conduction study showed motor axonal neuropathy with sensory sparing. A subsequent blood test revealed anti-GD1b IgG antibody positivity. He was diagnosed to have acute motor axonal neuropathy (AMAN) and treated with a course of intravenous immunoglobulin with slow improvement. This is probably the first AMAN with asymmetrical presentation mimicking stroke reported in the literature in detail. The anti-GD1b IgG antibody is also not commonly associated with AMAN.

Clival osteomyelitis and hypoglossal nerve palsy--rare complications of Lemierre's syndrome.

An increasingly reported entity, Lemierre's syndrome classically presents with a recent oropharyngeal infection, internal jugular vein thrombosis and the presence of anaerobic organisms such as Fusobacterium necrophorum. The authors report a normally fit and well 17-year-old boy who presented with severe sepsis following a 5-day history of a sore throat, myalgia and neck stiffness requiring intensive care admission. Blood cultures grew F. necrophorum and radiological investigations demonstrated left internal jugular vein, cavernous sinus and sigmoid sinus thrombus, left vertebral artery dissection and thrombus within the left internal carotid artery. Imaging also revealed two areas of acute ischaemia in the brain, consistent with septic emboli, skull base (clival) osteomyelitis and an extensive epidural abscess. The patient improved on meropenem and metronidazole and was warfarinised for his cavernous sinus thrombosis. He has an on-going left-sided hypoglossal (XIIth) nerve palsy.

Ulcerative colitis associated with isolated unilateral hypoglossal nerve palsy.

We herein describe a rare case of ulcerative colitis associated with unilateral hypoglossal nerve palsy. A 64-year-old woman developed severe active ulcerative colitis and was treated with prednisolone. The dose of oral prednisolone was reduced to 7.5 mg/day, following which the patient noticed slight dysphagia and a speech disturbance. She was diagnosed with unilateral hypoglossal nerve palsy, which was thought to be caused by mononeuritis. She was treated with intravenous methylprednisolone at a dose of 500 mg/day, which improved the neuropathy. When the neuropathy occurred, the patient was in a mildly active stage of ulcerative colitis. We concluded that the mononeuritis observed in the present case was likely an extraintestinal manifestation of ulcerative colitis.

Hypoglossal nerve palsy secondary to cervical spine involvement in rheumatoid arthritis: clinical and radiological features.

Isolated hypoglossal nerve (HN) palsy has been reported in a variety of disorders involving the cervical spine and/or skull base, however, unilateral HN palsy caused by rheumatoid arthritis (RA) has rarely been reported. We report herein an uncommon case of isolated HN palsy secondary to RA cervical spine involvement: pannus formation at the C1-C2 articulation, atlanto-axial subluxation, as well as, erosion of the right occipital condyle, lateral mass and anterior arch of C1. Pulse therapy with methylprednisolone followed by maintenance therapy with prednisone resulted in dramatic improvement. We also present the variety of diagnostic modalities helpful for the diagnosis and follow-up.

Subacute hypoglossal nerve paresis with internal carotid artery dissection.

OBJECTIVES: To describe a case of an isolated hypoglossal nerve palsy in a patient with a spontaneous internal carotid artery dissection (ICAD). This condition is a well-recognized cause of cerebral ischemic stroke in patients younger than 45 years of age. Isolated cranial nerve neuropathy is a rare presentation. More common manifestations include incomplete hemiparesis, hemicrania, Horner syndrome, cervical bruit, pulsatile tinnitus, and multiple cranial nerve palsies. METHODS: A comprehensive literature search (Ovid, MEDLINE) for the presentation, diagnostic evaluation, treatment, and outcome of patients with internal carotid artery dissection was performed. RESULTS: A 43-year-old man presented with a 3-week history of mild dysarthria. There was no history of craniocervical trauma. The physical examination revealed an isolated left hypoglossal nerve paresis. Magnetic resonance imaging and angiography findings were consistent with a left skull base ICAD. The patient was successfully treated with anticoagulation therapy. The current rate of cranial nerve involvement is estimated at 10% of all ICADs. This is the second report of isolated hypoglossal nerve palsy without hemicrania in a case of atraumatic ICAD. CONCLUSIONS: Patients with an ICAD infrequently present to the otolaryngologist because of its head and neck manifestations. It is crucial to recognize atypical findings and to perform an accurate and prompt diagnostic evaluation. The foundation of treatment is aggressive anticoagulation, with surgical or radiologic intervention reserved for cases demonstrating life-threatening progression.

[Isolated reversible unilateral paresis of hypoglossal nerve in tonsillitis--case report]. / Transitorische isolierte und unilaterale periphere Hypoglossusparese bei Tonsillitis--Falldarstellung.

We report on a 30-year-old patient with isolated, left-sided hypoglossal nerve palsy after uncomplicated, presumably streptococci-induced tonsillitis. Needle electromyography (EMG) of the tongue showed denervation changes in the muscles supplied by the left hypoglossal nerve. Cranial CT and MRI, CSF examination, Doppler sonography, visual evoked potential (VEP), and auditory evoked potential (AEP) showed no abnormalities and, in particular, no signs of the carotid artery dissection or brainstem lesion. The symptoms and signs resolved within a few weeks after penicillin V treatment. We suppose that the hypoglossal affection of the uncomplicated tonsillitis in this case was due to an aberrant position of the nerve.