Myorhythmia‐Like Dyskinesia Affecting the Face and Ear Associated With Anti–<i>N</i>‐Methyl‐<scp>d</scp>‐Aspartate Receptor Encephalitis

Camacho, Ana; Núñez, Noemí; Armangué, Thaís; Simón, Rogelio

doi:10.1002/mdc3.12300

Cited by 3 publications

(5 citation statements)

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“…Current understanding of the disorder, including treatment strategy and role of botulinum toxin, is largely gathered from case reports. [1][2][3][4][5] To date and to our knowledge, this is the first report on the use of ultrasonography to visualize spontaneous activity of the various muscles involved. Ultrasonography was used to confirm involvement of both the auricularis posterior and superior muscles without subjecting the patient to a potentially uncomfortable electromyographic examination.…”

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confidence: 88%

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Ultrasonography Evaluation of Bilateral Focal Auricular Dyskinesia

Prasanna

Wee

2021

JAMA Neurol

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An otherwise healthy woman inherlate20spresentedwitha1-week history of almost continuous bilateral ear twitching. Onset was spontaneous. There was no previous exposure to neuroleptics nor history of neuropsychiatric disorders. The ear movements were not within voluntary control nor distractible under cognitive load. These movements caused significant distress including poor sleep and headaches.Shedeniedfeelingstressedoranxious.Themovementsstopped during sleep. She denied any tinnitus.Examination revealed semirhythmic, asymmetrical movement of bilateral ears and the frontalis muscle. The character of muscle movement was observed on ultrasonography. Muscle contraction and shortening generally began as quick and jerky movements, and the degree of contraction ranged from complete to partial. Muscle relaxation and lengthening occurredatvariablespeed,fromquickandjerky to slow and sinuous. Neurologic examination results were otherwise normal with no tremors or dyskinesias in thelimbs.TheVideodemonstratestheclinicalpresentationandmuscle movements visualized under ultrasonography examination.Full blood cell count and inflammatory markers were within normal limits. Electrolyte levels including calcium, magnesium, and phosphate were normal. Magnetic resonance imaging results of the brain were normal.The patient was diagnosed as having auricular dyskinesia with no obvious precipitating factor and started taking baclofen, 10 mg, 3 times adaywithnoimprovement.Thiswaschangedtoclonazepam,0.25mg, 3 times a day. She was able to sleep better with clonazepam, but it had no effect on the frequency or intensity of the twitching.The patient was offered a trial of botulinum toxin injection in view of her poor response to oral medications. Ultrasonography (Aixplorer Mach 30 with a high-frequency linear transducer SLH20-6 using musculoskeletal preset [resolution mode; depth, 1 cm] with focus set at the structure of interest) was used for muscle examination. Bilateral auricularis superior and posterior muscles were injected with 10 units of onabotulinum toxin type A. A further 10 units were injected into the frontal bellies of the occipitofrontalis muscle bilaterally in the area with the most active muscle movements seen under ultrasonography. The Figure shows a pictorial representation of the muscles. At follow-up 2 weeks later, all spontaneous movements had ceased. She did not experience any adverse events apart from worsening of the existing headache for a few days.

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confidence: 88%

“…Auricular dyskinesia is uncommon. Current understanding of the disorder, including treatment strategy and role of botulinum toxin, is largely gathered from case reports . To date and to our knowledge, this is the first report on the use of ultrasonography to visualize spontaneous activity of the various muscles involved.…”

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confidence: 99%

Ultrasonography Evaluation of Bilateral Focal Auricular Dyskinesia

Prasanna

Wee

2021

JAMA Neurol

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“…However, autoimmune disease also may give rise to myorhythmia, particularly with IgLON5 antibodies, 61,62 but also NMDA receptor and KLHL11 encephalitis. 63 Other, difficult-to-classify movement disorder presentations of autoimmune disease are often the result of a combination of psychiatric symptoms (eg, with catatonia, clonic or tonic perseveration) with peripheral movement disorder mimics (eg, peripheral nerve hyperexcitability as in fibrillary chorea) or of an unusual phenomenology of the movement disorder itself. Examples of this notion are abdominal dyskinesia due to neuromyotonia reported in IgLON5 disease, 64 trunk flexion or abdominal wall contractions superficially resembling psychogenic propriospinal myoclonus in CASPR2 disease, 65 or movement disorders defying the classic movement disorder terminology in NDMA receptor or GABA A receptor encephalitis.…”

Section: Key Pointsmentioning

confidence: 99%

“…Myorhythmia, particularly oculomasticatory myorhythmia with vertical gaze palsy, is a classic presentation of neurologic Whipple disease. However, autoimmune disease also may give rise to myorhythmia, particularly with IgLON5 antibodies, 61,62 but also NMDA receptor and KLHL11 encephalitis 63 …”

Section: Tremor Myorhythmia and Other Syndromesmentioning

confidence: 99%

Autoimmune Movement Disorders

Balint

2024

CONTINUUM: Lifelong Learning in Neurology

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OBJECTIVE This article reviews the clinical and antibody spectrum of autoimmune cerebellar ataxia and other autoimmune movement disorders. It highlights characteristic phenotypes and red flags to the diagnosis and how these rare, but treatable, disorders are integrated into a differential diagnosis. LATEST DEVELOPMENTS An increasing number of neuronal antibodies have been identified in patients with cerebellar ataxia, for example, against Kelch-like protein 11 (KLHL11), seizure-related 6 homolog–like 2, septin-3 and septin-5, or tripartite motif containing protein 9 (TRIM9), TRIM46, and TRIM67. Ig-like cell adhesion molecule 5 (IgLON5) antibody–associated syndromes have emerged as an important alternative diagnostic consideration to various neurodegenerative diseases such as Huntington disease or atypical parkinsonism. Opsoclonus-myoclonus syndrome emerged as the most relevant parainfectious movement disorder related to severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). ESSENTIAL POINTS Autoimmune cerebellar ataxia and other autoimmune movement disorders encompass a broad spectrum of different clinical syndromes, antibodies, and immunopathophysiologic mechanisms. Clinical acumen is key to identifying the cases that should undergo testing for neuronal antibodies. Given the overlap between phenotypes and antibodies, panel testing in serum and CSF is recommended.

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“…It has been frequently reported as oculo-masticatory myorhythmia in the context of Whipple's disease, caused by the infection of the central nervous system by Trophyrema whipplei ( 140 ). Other conditions where myorhythmia has been reported are stroke ( 139 ), anti-NMDA encephalitis ( 141 , 142 ), anti-IgLON5 disease ( 143 ), interferon alpha-2a use ( 144 ), Hashimoto encephalopathy ( 145 ), and X-linked dystonia-parkinsonism ( 146 ). As myorhythmia is often associated with conditions that are potentially treatable, it is important to be familiar with this phenomenology and its differential diagnoses.…”

Section: Tremor In the Setting Additional Neurological Featuresmentioning

confidence: 99%

Tremor Syndromes: An Updated Review

Lenka

Jankovic

2021

Front. Neurol.

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Tremor is the most commonly encountered movement disorder in clinical practice. A wide range of pathologies may manifest with tremor either as a presenting or predominant symptom. Considering the marked etiological and phenomenological heterogeneity, it would be desirable to develop a classification of tremors that reflects their underlying pathophysiology. The tremor task force of the International Parkinson Disease and Movement Disorders Society has worked toward this goal and proposed a new classification system. This system has remained a prime topic of scientific communications on tremor in recent times. The new classification is based on two axes: 1. based on the clinical features, history, and tremor characteristics and 2. based on the etiology of tremor. In this article, we discuss the key aspects of the new classification, review various tremor syndromes, highlight some of the controversies in the field of tremor, and share the potential future perspectives.

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Myorhythmia‐Like Dyskinesia Affecting the Face and Ear Associated With Anti–N‐Methyl‐d‐Aspartate Receptor Encephalitis

Cited by 3 publications

References 10 publications

Ultrasonography Evaluation of Bilateral Focal Auricular Dyskinesia

Ultrasonography Evaluation of Bilateral Focal Auricular Dyskinesia

Autoimmune Movement Disorders

Tremor Syndromes: An Updated Review

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