Involuntary Tongue Movements Research Articles

Buerger’s disease with cerebral involvement in a middle-aged woman with diabetes mellitus. A case report

Introduction: Buerger’s disease (BD) generally affects men, young people, and smokers, but it can also affect women. Its incidence is rare in Latin America. Case report: A 40-year-old Colombian woman, active smoker and user of psychoactive substances, attended the emergency department of a tertiary care center due to symptoms of 3 days consisting of retraction of the corner of the mouth, drooling, and involuntary tongue movements. The patient, who had a history of uncontrolled diabetes and recent acute ischemia of the right upper limb due to acute thrombosis, required surgical management and subsequent use of oral anticoagulation. She later developed necrotic changes in the distal phalanges of the right hand that required ablative therapy. Since age, sex and limb involvement were not typical for BD, collagenosis, vasculitis or thrombophilia were ruled out, but after excluding these disorders, BD with atypical features was considered. The patient was discharged with oral anticoagulation, aspirin, combined analgesia, physiotherapy, and recommendation for smoking cessation. Conclusions: Age, sex, smoking and comorbidities such as diabetes are risk factors for BD. Imaging and histopathology are the gold standard for the definitive diagnosis of this entity. Multidisciplinary management, lifestyle changes, smoking cessation, pain control, good wound healing and social support are key aspects for better clinical outcomes in patients with BD.

Restless tongue: Lingual Tardive Dyskinesia – A rare case report

Tardive Dyskinesia is a movement disorder characterized by involuntary repetitive body movements, like chewing motions, cheek puffing, tongue protrusion and lip pursing. These symptoms appear during sleep and/or wakefulness. Report of involuntary movements of tongue is very rare, with a prevalence of only 15%–20%. Risk factors include old age, female gender, and patients receiving drugs with anti-dopaminergic activity, for long term. We report a case of 62 years old male patient with a long history of antidepressants and mood stabilizers, who presented with involuntary tongue movements. The patient was treated by altering the dose and discontinuation of a few medicines, which resulted in a slight decrease in the frequency of the movement. The case is discussed here to spread awareness and vigilance about this condition, in order to aid in early diagnosis and management, to avoid negative impacts on psychologic health and quality of life of the patient.

Huntington disease-like phenotype in a patient with ANO3 mutation

A 71-year-old previously well white British female developed progressive involuntary tongue movements over one year, resulting in eating difficulty and 10 kg weight loss. She had also noted involuntary perioral, facial and distal limb movements beginning 18 months earlier. These had progressively worsened. In the 3 years prior to presentation, she reported subjective memory decline, word finding difficulty and depressed mood, which improved with mirtazapine 30 mg once daily. She had no history of neuroleptic exposure.

Oromandibular dystonia, mental distress and oro-facial dysfunction-A follow-up 8-10years after start of treatment with botulinum toxin.

Oromandibular dystonia (OMD) with involuntary jaw and tongue movements may be misdiagnosed as temporomandibular disorders (TMD) and because of the complex muscle activity and involvement of several small muscles, OMD is also considered difficult to treat. The aim was to evaluate OMD in patients 8-10years after start of treatment with botulinum toxin (BoNT) by self-reported and standardised global scales and questionnaires. Of 21 previously reported patients with OMD, 14 responded to a mail health questionnaire to describe the disease course and treatment effect as well as the overall impact of OMD by a visual analogue scale (VAS), the Patient Health Questionnaire (PHQ) for depression and anxiety, and the Jaw Functional Limitation Scale (JFLS). The results were analysed with non-parametric statistical analysis (Wilcoxon matched-pairs test and Spearman's rank-order correlations). The OMD was still present in 13 patients. In nine patients, the BoNT treatment had continued as monotherapy or combined with oral medication. VAS for OMD was significantly reduced (P<0.04) over the years, and most patients felt improvement from the treatment. However, the patients had still some functional limitations, typically regarding jaw mobility and communication, and both JFLS and mental distress (PHQ) were significantly correlated with the OMD VAS (rS 0.77 and 0.74). The results showed marked reduction of the experienced OMD with treatment and over time, and also stressed similarities between OMD and TMD. Both dentists and neurologists should be aware of this overlap and reduce misdiagnosis by applying an interdisciplinary approach.

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features.

Lingual dystonia is a debilitating type of oromandibular dystonia characterized by involuntary, often task-specific, contractions of the tongue muscle activated by speaking or eating. Botulinum neurotoxin (BoNT) has been used to treat lingual dystonia; however, it is known to cause serious complications, such as dysphasia and aspiration. The purpose of this study was to evaluate the efficacy and adverse effects of individualized BoNT therapy for lingual dystonia. One-hundred-and-seventy-two patients (102 females and 70 males, mean age: 46.2 years) with lingual dystonia were classified into four subtypes based on symptoms of involuntary tongue movements: protrusion (68.6%), retraction (16.9%), curling (7.6%), and laterotrusion (7.0%). Patients were treated with BoNT injection into the genioglossus and/or intrinsic muscles via individualized submandibular and/or intraoral routes. Results were compared before and after BoNT therapy. Botulinum neurotoxin was injected in 136 patients (mean: 4.8 injections). Clinical sub-scores (mastication, speech, pain, and discomfort) in a disease-specific rating scale were reduced significantly (p < 0.001) after administration. Comprehensive improvement after BoNT injection, assessed using the rating scale, was 77.6%. The curling type (81.9%) showed the greatest improvement, while the retraction type showed the least improvement (67.9%). Mild and transient dysphasia occurred in 12.5% of patients (3.7% of total injections) but disappeared spontaneously within several days to two weeks. No serious side effects were observed. With careful diagnosis of subtypes and a detailed understanding of lingual muscle anatomy, individualized BoNT injection into dystonic lingual muscles can be effective and safe.

New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.

Objective:To determine the causative pathogen and investigate the effective treatment of a new type of encephalomyelitis with an unknown pathogen in Japan and report the preliminary ultrastructural and genomic characterization of the causative agent.Methods:From 2005 to 2012, we treated 4 Japanese patients with geographic clustering and comparable clinical features, serum/CSF cytology, and radiologic findings. Brain biopsy was conducted in all patients to analyze neuropathologic changes by histology, and electron microscopy was applied to reveal the features of the putative pathogen. Genomic DNA was obtained from the affected brain tissues and CSF, and an unbiased high-throughput sequencing approach was used to screen for specific genomic sequences indicative of the pathogen origin.Results:All patients exhibited progressive dementia with involuntary tongue movements. Cytologic examination of CSF revealed elevated mononuclear cells. Abnormal MRI signals were observed in temporal lobes, subcortical white matter, and spinal cord. Biopsied brain tissue exhibited aggregated periodic acid-Schiff–positive macrophages and 2–7 μm diameter round/oval bodies without nuclei or cell walls scattered around the vessels. Unbiased high-throughput sequencing identified more than 100 archaea-specific DNA fragments. All patients were responsive to trimethoprim/sulfamethoxazole (TMP-SMX) plus corticosteroid therapy.Conclusions:We report 4 cases of encephalomyelitis due to an unknown pathogen. On the basis of ultrastructural and genomic studies, we propose a new disease entity resulting from a causative pathogen having archaeal features. TMP-SMX therapy was effective against this new type of encephalomyelitis.

Co-occurrence of Dystonic and Dyskinetic Tongue Movements with Oral Apraxia in Post-regression Dysphagia in Classical Rett Syndrome Years of Life 1 Through 5.

We do not know the natural history of dysphagia in classical Rett syndrome (RTT) by stage or age. This study investigated swallowing physiology in 23 females ages 1:7 to 5:8 (years, months) with classical Rett syndrome to determine common and distinguishing features of dysphagia in post-regression early Pseudostationary Stage III. In-depth analysis of videofluoroscopic swallowing studies (VFSS) found dysmotility of oral stage events across subjects implicating oral apraxia. Impaired motility was further compromised by recurrent dystonic and dyskinetic movements that co-occurred with oral apraxia during oral ingestion in 78% (n=18) of the subjects with RTT. Of this group, 44% displayed rocking and/or rolling lingual pattern, 56% had recurrent oral tongue retroflexions, and/or elevated posturing of the tongue tip, and, 72% displayed multi-wave oropharyngeal transfer pattern. The proportion of subjects whose swallowing motility was disrupted by aberrant involuntary tongue movements did not differ significantly between bolus types (liquid, puree, and solid) trialed. Liquid ingestion was significantly more efficient in subjects using bottles with nipples than their counterparts who used spouted or straw cups. Dystonic and dyskinetic tongue movements disrupted liquid ingestion in subjects using cups with spouts or straws significantly more than those using bottles. Analysis of food ingestion revealed that significantly more subjects were able to orally form, transport, and transfer a puree bolus into the pharynx than they were a solid bolus. A significantly larger number of subjects aspirated and penetrated liquid than they did puree or solid. No significant relationship was found between subjects with airway contamination and those with dystonic and dyskinetic tongue movements. Subjects' rocking and rolling lingual patterns were consistent with those evidenced in adults with Parkinson's disease. Subjects' tongue retroflexions were classified as provisionally unique to RTT. VFSS pre-planning, fluoroscopic procedures, and therapeutic strategies specific to this specialty population were derived.

Motor neuron disorder with tongue spasms due to pyrethroid insecticide toxicity

Pyrethroids are widely used insecticides because of their high potency and relatively low toxicity to mammals.1 Symptoms of acute pyrethroid neurotoxicity in humans include numbness, dizziness, muscle fasciculations, and, in more severe cases, hypersalivation, seizures, and coma.1 Recently, 2 cases of partly or completely irreversible motor neuron disorder (MND) secondary to chronic pyrethroid inhalation were reported.2,3 We report a case of reversible MND and involuntary tongue movements (ITMs) secondary to transient pyrethroid overexposure. ### Case report. A 60-year-old farmer presented a 3-month history of slurred speech due to ITMs, preceded by a burning sensation of the tongue and palate associated with facial paresthesias. ITMs were continuous without any clear triggering or alleviating factor. The patient denied traumatic event, infectious episode, or neuroleptic intake. Family history was unrevealing. He reported a similar episode 2 years earlier that resolved spontaneously over a few months. He admitted having used type II pyrethroid insecticides (λ-cyhalothrin and cypermethrin) disregarding the recommended precautions. He regularly manipulated (about 10 times per year for …

Isolated continuous rhythmic involuntary tongue movements following a pontine infarct

Isolated involuntary tongue movements are rare and poorly understood. The anatomical substrate and pathogenesis underlying involuntary tongue movements remain elusive. We describe a patient who developed isolated continuous rhythmic involuntary tongue movements after pontine infarct without evidence of hypertrophy of inferior olivary nucleus on follow-up magnetic resonance image. We discuss the rhythmic involuntary tongue movements as a prototype of involuntary hyperkinetic movement released by a central pacemaker.

Neostriatal stimulation activates tongue-protruder muscle, but not tongue-retractor or facial muscles: an electrical and chemical microstimulation study in rats

Whether electrical microstimulation of a rat’s striatal jaw region (SJR) in fact induced tongue or facial muscle activity in addition to jaw muscle activity was tested. Microstimulation of SJR-evoked EMG activity in a jaw-opener (anterior digastricus) and tongue-protruder (genioglossus). No activity was evoked in jaw-closers (temporalis or masseter), tongue-retractor (hyoglossus) or in facial muscles. In addition, the EMG effect could still be induced after extensive ablation of the neocortex; it was reproduced by microinjection of 50 mM kainic acid into SJR. The effective sites were histologically localized to a small central striatal region adjoining the anterior commissure. These findings may be of considerable value in understanding the striatal mechanism of orolingual dyskinesia involving involuntary jaw and tongue movements.

Source density analysis of scalp potentials during evaluated action. I. Coronal distribution.

The distribution of source densities (the "Laplacean") of event related potentials (ERPs) over the scalp during a goal-directed task has been derived by a novel method which presents seven samples in a hexagonal array, using an economical computing technique that affords much freedom from artefactual contamination. The task had four phases: waiting for a ball to appear, observing its coordinates and estimating action needed to get it through a goal-mouth, acting and awaiting the outcome, and finally evaluating the outcome as success or failure. Different phases of the task were characterized by significantly different distributions of sources. Failures were distinguished from success to consistent features of the last phase of the ERP. All locations within the reference hexagon (spanning a circle of approximately 12 cm diameter on the scalp) showed the latter features. In control experiments, the effects of response mode and of different cognitive interpretations of the input/output situation were studied. A check showed that with monopolar recording involuntary tongue movements can totally vitiate conclusions in such investigations. The Laplacean derivations proved relatively free of such artefactual contamination.