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Movement Disorders Clinical Practice Feb 2021
PubMed: 33816652
DOI: 10.1002/mdc3.13138 -
BMC Ophthalmology Dec 2020We report a case of Meige's syndrome induced by an atypical antipsychotic (blonanserin) that presented with refractory dry eye disease.
BACKGROUND
We report a case of Meige's syndrome induced by an atypical antipsychotic (blonanserin) that presented with refractory dry eye disease.
CASE PRESENTATION
A 37-year-old woman with a 6-month history of foreign body sensation in the eyes and difficulty in opening her eyes was treated at a local clinic for dry eye disease. Despite this treatment, her symptoms did not improve and she was transferred to our attention. Our assessment revealed involuntary movements of her eyelids accompanied by repetitive pursing of her lips. She had been undergoing treatment with blonanserin for 5 years for schizophrenia. She was diagnosed with drug-induced Meige's syndrome after a psychiatric and neurological consultation. After a 2-month gradual dose reduction and discontinuing blonanserin, involuntary movements of the eyelids with oromandibular dystonia were resolved. Three months after discontinuing blonanserin, there was no recurrence of symptoms, and she had no exacerbation of psychotic symptoms.
CONCLUSIONS
In patients with refractory dry eye disease, especially those with involuntary movements of the eyelids with oromandibular dystonia, it is important to ask about their psychotropic medications and to consider the possibility of drug-induced Meige's syndrome and discontinuation of medications, if possible.
Topics: Adult; Antipsychotic Agents; Dry Eye Syndromes; Female; Humans; Meige Syndrome
PubMed: 33267850
DOI: 10.1186/s12886-020-01738-w -
Cureus Sep 2020Meige syndrome, also known as blepharospasm-oromandibular dystonia, is a neurological movement disorder that involves the involuntary muscle contractions of the eyes,...
Meige syndrome, also known as blepharospasm-oromandibular dystonia, is a neurological movement disorder that involves the involuntary muscle contractions of the eyes, mouth, tongue, and jaw. It is often associated with other disorders, such as Parkinson's disease. We describe a case of an 87-year-old man with Meige syndrome who was successfully treated with oral baclofen.
PubMed: 33101815
DOI: 10.7759/cureus.10570 -
Scientific Reports Sep 2020To investigate the changes and clinical significance of brain structural abnormalities in patients with Meige syndrome and related depressive symptoms. We...
To investigate the changes and clinical significance of brain structural abnormalities in patients with Meige syndrome and related depressive symptoms. We retrospectively analysed clinical data, imaging examinations, and Hamilton Depression Rating scale scores in 46 patients with Meige syndrome from January 2017 to January 2019. We compared the Meige syndrome group with the healthy control group, and the definite depression group with the non-definite depression group. Voxel-based morphometry (VBM) was used to compare grey matter (GM) volumes. We conducted two-sample t-tests corrected for subject age and gender. We tested at a level of significance of p < 0.001 with a false discovery rate (FDR) correction. VBM demonstrated decreased GM volume (p < 0.001 and cluster size > 50 voxels) in the left hemisphere in the middle frontal orbital gyrus, temporal pole (superior temporal gyrus) and insula and in the right hemisphere in the temporal pole (middle temporal gyrus), precuneus, inferior parietal, inferior temporal and olfactory cortices in the Meige syndrome group. Comparing VBM-MRI measures in Meige syndrome patients with and without depression, decreased GM volume was found in the left hemisphere in the cuneus and hippocampus and in the right hemisphere in the angular gyrus, middle frontal gyrus and middle occipital gyrus in the definite depression group. Unlike other dystonia studies that have suggested an involvement of the basal ganglia and motor cortex in the pathophysiology of the disorder , we believe that the precuneus is involved in the development of Meige syndrome. Additionally, our findings suggest that the hippocampus plays a role in the pathogenesis of depression in patients with Meige syndrome.
Topics: Aged; Female; Gray Matter; Humans; Magnetic Resonance Imaging; Male; Meige Syndrome; Middle Aged; Motor Cortex; Retrospective Studies
PubMed: 32884000
DOI: 10.1038/s41598-020-71479-9 -
Frontiers in Neurology 2020The effect of deep brain stimulation (DBS) on swallowing function in movement disorders is unclear. Here, we systematically reviewed this topic by searching keywords...
The effect of deep brain stimulation (DBS) on swallowing function in movement disorders is unclear. Here, we systematically reviewed this topic by searching keywords following PICOS strategy of problem (swallowing or swallow or dysphagia or aspiration) and intervention (deep brain stimulation, or DBS) in the PubMed and Web of Science in English in April 2020, with comparators [subthalamic nucleus (STN), globus pallidus interna (GPi), ventralis intermedius, (ViM), post-subthalamic area, or caudal zona incerta (PSA/cZi); ON/OFF DBS state/settings, ON/OFF medication state, Parkinson's disease (PD), dystonia, tremor], outcomes (swallowing function measures, subjective/objective) and study types (good quality original studies) in mind. We found that STN DBS at usual high-frequency stimulation could have beneficial effect (more so on subjective measures and/or OFF medication), no effect, or detrimental effect (more so on objective measures and/or ON medication) on swallowing function in patients with PD, while low-frequency stimulation (LFS) could have beneficial effect on swallowing function in patients with freezing of gait. GPi DBS could have a beneficial effect (regardless of medication state and outcome measures) or no effect, but no detrimental effect, on swallowing function in PD. GPi DBS also has beneficial effects on swallowing function in majority of the studies on Meige syndrome but not in other diseases with dystonia. PSA/cZi DBS rarely has detrimental effect on swallowing functions in patients with PD or tremor. There is limited information on ViM to assess. Information on swallowing function by DBS remains limited. Well-designed studies and direct comparison of targets are further needed.
PubMed: 32765388
DOI: 10.3389/fneur.2020.00547 -
Revista de Neurologia Jul 2020Dyskinesia of the ADCY5 mutation is a rare movement-onset disorder in childhood. It is characterized by isolated chorea movements or associated with myoclonus and... (Review)
Review
INTRODUCTION
Dyskinesia of the ADCY5 mutation is a rare movement-onset disorder in childhood. It is characterized by isolated chorea movements or associated with myoclonus and dystonia affecting the limbs, neck and face. The low number of patients and families still does not allow an adequate genotype-phenotype relationship.
AIMS
The case of a child with movement disorders of early onset is presented in a family with three generations of affected members. An updated review of the casuistry and management of this rare disease is made.
CASE REPORT
A 6-year-old boy referred for language delay and hyperactivity. After six months of follow-up he begins to show chorea movements of predominantly facial and limb roots, especially when waking up. At one year of follow-up, generalized chorea at rest with orofacial involvement and awkward gait begins to show. His family history includes his mother, grandfather, maternal uncle and cousin, who were diagnosed with Meige's syndrome (oromandibular dystonia and periorbital muscles) with choreiform-like movement disorders without affiliation since childhood. The brain study by MRI showed no alterations. A clinical exome targeting movement disorders was performed that discovered the pathogenic mutation in the ADCY5 gene causing autosomal familial dyskinesia.
CONCLUSION
The c.1126G>A p.A376T mutation shows a natural history with a non-progressive clinical phenotype in three generations of affected members, with childhood debut and response to guanfacine treatment.
Topics: Adenylyl Cyclases; Amino Acid Substitution; Attention Deficit Disorder with Hyperactivity; Child; Developmental Disabilities; Drug Resistance; Female; Guanfacine; Humans; Language Development Disorders; Levetiracetam; Male; Meige Syndrome; Movement Disorders; Mutation, Missense; Pedigree; Point Mutation
PubMed: 32627162
DOI: 10.33588/rn.7102.2020154 -
Toxins Apr 2020Blepharospasm and oromandibular dystonia are focal dystonias characterized by involuntary and often patterned, repetitive muscle contractions. There is a long history of... (Review)
Review
Blepharospasm and oromandibular dystonia are focal dystonias characterized by involuntary and often patterned, repetitive muscle contractions. There is a long history of medical and surgical therapies, with the current first-line therapy, botulinum neurotoxin (BoNT), becoming standard of care in 1989. This comprehensive review utilized MEDLINE and PubMed and provides an overview of the history of these focal dystonias, BoNT, and the use of toxin to treat them. We present the levels of clinical evidence for each toxin for both, focal dystonias and offer guidance for muscle and site selection as well as dosing.
Topics: Blepharospasm; Botulinum Toxins; Dystonic Disorders; Humans; Mandibular Diseases; Muscular Diseases; Neuromuscular Agents
PubMed: 32331272
DOI: 10.3390/toxins12040269 -
Neurologic Clinics May 2020The dystonias are a large and heterogenous group of disorders characterized by excessive muscle contractions leading to abnormal postures and/or repetitive movements.... (Review)
Review
The dystonias are a large and heterogenous group of disorders characterized by excessive muscle contractions leading to abnormal postures and/or repetitive movements. Their clinical manifestations vary widely, and there are many potential causes. Despite the heterogeneity, helpful treatments are available for the vast majority of patients. Symptom-based therapies include oral medications, botulinum toxins, and surgical interventions. For some subtypes of dystonia, specific mechanism-based treatments are available. Advances in understanding the biological basis for many types of dystonia have led to numerous recent clinical trials, so additional treatments are likely to become available in the very near future.
Topics: Dystonia; Humans
PubMed: 32279713
DOI: 10.1016/j.ncl.2020.01.003 -
Neuropsychopharmacology Reports Dec 2019The main symptoms of Meige's syndrome are involuntary eye blinking with muddled speech and uncontrollable contraction of the platysma muscle characterized by segmental,...
The main symptoms of Meige's syndrome are involuntary eye blinking with muddled speech and uncontrollable contraction of the platysma muscle characterized by segmental, primarily oromandibular, dystonia (hyperkinesia). It can also develop after long-term medication of first- and second-generation antipsychotics. Here, we report the case of a Japanese female schizophrenic patient comorbid with Meige's syndrome and hyperthyroidism. We discuss the relationship between the three diseases, that is, schizophrenia, Meige's syndrome, and hyperthyroidism. Our intention is to consider the important role of the cerebral basal ganglia, where little attention has been given in regard to schizophrenia and Meige's syndrome. A part of this article was presented in a poster section at the joint congress of the 28th Annual Meeting of the Japanese Society of Clinical Neuropsychopharmacology and the 48th Annual Meeting of the Japanese Society of Neuropsychopharmacology held in 2018.
Topics: Female; Humans; Hyperthyroidism; Japan; Meige Syndrome; Middle Aged; Schizophrenia
PubMed: 31743613
DOI: 10.1002/npr2.12081 -
Journal of Thoracic Oncology : Official... Nov 2019
Topics: Adenocarcinoma of Lung; Aged; Fatal Outcome; Female; Gene Rearrangement; Humans; Lung Neoplasms; Meige Syndrome; Proto-Oncogene Proteins c-ret
PubMed: 31668323
DOI: 10.1016/j.jtho.2019.06.008