Lingual Hyperkinesia as An Initial Manifestation of WernickeâÂÂsEncephalopathy: Evidence for Localization of Involuntary HyperkineticMovement of the Tongue

Background: Movement disorders caused by Wernicke’s encephalopathy (WE) are very rarely reported, and involuntary hyperkinesia of the tongue as an initial manifestation of WE have not yet been reported. The study aimed to investigate the path mechanism and localization of involuntary lingual movement symptoms. Methods: We present a patient with lingual hyperkinesia as an initial manifestation of WE, who had lesions of the periaqueductal area of the midbrain tectum and bilaterally medial thalami in Brain MRI. Patient’s lingual symptoms were observed and analyzed sequentially for localization of involuntary hyperkinetic movement of the tongue. Results: Lingual hyperkinesia symptoms of the patient diagnosed in WE, occurred in advance and were more earlier improved by thiamine treatment before other brainstem symptoms. Conclusion: The clinical and neuro-radiological results discussed here may provide support for the localization of lingual hyperkinetic movement disorders. In addition to brainstem lesion, thalamic lesion should be considered in cases of acute or subacute-onset, involuntary hyperkinetic movement of the tongue.

[1]  Dokyung Lee,et al.  Isolated Glossoplegia in Capsular Infarction , 2016, Canadian Journal of Neurological Sciences / Journal Canadien des Sciences Neurologiques.

[2]  M. Hallett,et al.  Isolated Lingual Dyskinesia in Multiple Sclerosis. , 2015, JAMA neurology.

[3]  D. Cordato,et al.  Complete tongue protrusion palsy due to subsequent cryptogenic cortical strokes , 2015, BMJ Case Reports.

[4]  A. Manto,et al.  MR Imaging Findings in Alcoholic and Nonalcoholic Acute Wernicke's Encephalopathy: A Review , 2014, BioMed research international.

[5]  H. Hanagasi,et al.  Orolingual dyskinesia and involuntary neck movements caused by phenytoin intoxication. , 2013, The Journal of neuropsychiatry and clinical neurosciences.

[6]  G. Bråthen,et al.  EFNS guidelines for diagnosis, therapy and prevention of Wernicke encephalopathy , 2010, European journal of neurology.

[7]  C. Civardi,et al.  Isolated continuous lingual myoclonus: unusual presentation of amyotrophic lateral sclerosis , 2010, Movement disorders : official journal of the Movement Disorder Society.

[8]  Hye-Jin Kim,et al.  Lingual dystonia as a manifestation of thalamic infarction , 2009, Movement disorders : official journal of the Movement Disorder Society.

[9]  Phil Hyu Lee,et al.  Dystonic hand tremor in a patient with Wernicke encephalopathy. , 2009, Parkinsonism & related disorders.

[10]  Chieh-Hsun Lee,et al.  Episodic tongue hyperkinesias and alternating limb movements associated with basilar artery ischemia , 2009, Movement disorders : official journal of the Movement Disorder Society.

[11]  S. Yoon,et al.  Glossoplegia in a small cortical infarction , 2009, BMJ Case Reports.

[12]  N. Pipitone,et al.  Neuroimaging findings in acute Wernicke's encephalopathy: review of the literature. , 2009, AJR. American journal of roentgenology.

[13]  A. Espay,et al.  Teaching NeuroImage: Oculomasticatory myorhythmia , 2008, Neurology.

[14]  Y. Lo,et al.  Cortico-hypoglossal and corticospinal conduction abnormality in Bickerstaff's brainstem encephalitis , 2007, Clinical Neurology and Neurosurgery.

[15]  A. Serra,et al.  Wernicke's encephalopathy: new clinical settings and recent advances in diagnosis and management , 2007, The Lancet Neurology.

[16]  B. Yoon,et al.  Upbeat Nystagmus: Clinicoanatomical Correlations in 15 Patients , 2006, Journal of clinical neurology.

[17]  P. Martinelli,et al.  Drug-induced tremor of the tongue , 1989, The Italian Journal of Neurological Sciences.

[18]  Phil Hyu Lee,et al.  Isolated continuous rhythmic involuntary tongue movements following a pontine infarct. , 2005, Parkinsonism & related disorders.

[19]  Jae-Hong Lee,et al.  Isolated tongue tremor after gamma knife radiosurgery for acoustic schwannoma , 2005, Movement disorders : official journal of the Movement Disorder Society.

[20]  Kátia Lin,et al.  Tongue tremor in a patient with coma after electrical injury , 2003, Movement disorders : official journal of the Movement Disorder Society.

[21]  Y. Lo,et al.  Corticobulbar dysfunction in the Miller Fisher syndrome , 2003, Clinical Neurology and Neurosurgery.

[22]  M. Hallett,et al.  Analogous Corticocortical Inhibition and Facilitation in Ipsilateral and Contralateral Human Motor Cortex Representations of the Tongue , 2001, Journal of clinical neurophysiology : official publication of the American Electroencephalographic Society.

[23]  H. Przuntek,et al.  Episodic undulating hyperkinesias of the tongue associated with brainstem ischemia , 1997, Movement disorders : official journal of the Movement Disorder Society.

[24]  A. Avila,et al.  Isolated lingual myoclonus associated with an Arnold-Chiari malformation. , 1994, Journal of neurology, neurosurgery, and psychiatry.

[25]  R. Moodley,et al.  Dystonia and choreo-athetosis in Wernicke's encephalopathy. A case report. , 1989, South African medical journal = Suid-Afrikaanse tydskrif vir geneeskunde.

[26]  P. Rudge,et al.  Primary position upbeating nystagmus. A variety of central positional nystagmus. , 1983, Brain : a journal of neurology.

[27]  B. Jabbari,et al.  Paroxysmal, rhythmic lingual movements and chronic epilepsy , 1981, Neurology.