Contemporary encephalitis lethargica presenting with agitated catatonia, stereotypy, and dystonia-parkinsonism.
Identifieur interne : 002506 ( PubMed/Curation ); précédent : 002505; suivant : 002507Contemporary encephalitis lethargica presenting with agitated catatonia, stereotypy, and dystonia-parkinsonism.
Auteurs : Russell C. Dale [Australie] ; Richard Webster ; Deepak GillSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2007.
English descriptors
- KwdEn :
- Adolescent, Catatonia (diagnosis), Catatonia (etiology), Catatonia (therapy), Child, Dopamine (physiology), Dystonia (diagnosis), Dystonia (etiology), Dystonia (therapy), Humans, Intensive Care, Male, Parkinson Disease, Postencephalitic (complications), Parkinson Disease, Postencephalitic (diagnosis), Parkinson Disease, Postencephalitic (therapy), Recovery of Function, Stereotypic Movement Disorder (diagnosis), Stereotypic Movement Disorder (etiology), Stereotypic Movement Disorder (therapy).
- MESH :
- chemical , physiology : Dopamine.
- complications : Parkinson Disease, Postencephalitic.
- diagnosis : Catatonia, Dystonia, Parkinson Disease, Postencephalitic, Stereotypic Movement Disorder.
- etiology : Catatonia, Dystonia, Stereotypic Movement Disorder.
- therapy : Catatonia, Dystonia, Parkinson Disease, Postencephalitic, Stereotypic Movement Disorder.
- Adolescent, Child, Humans, Intensive Care, Male, Recovery of Function.
Abstract
Encephalitis lethargica (EL) syndrome was classically described by Von Economo and has somnolent-ophthalmoplegic, hyperkinetic, and amyostatic-akinetic forms. We describe 2 recent cases of EL characterized by an acute encephalitis with mixed movement disorders (dystonia-Parkinsonism plus stereotypy) and psychiatric disorders (agitated catatonia, coprolalia, and echo phenomena). Both patients suffered concurrent hyperkinetic and Parkinsonian features resulting in therapeutic challenges. Bradykinetic features responded to dopamine replacement therapy and both patients also had adverse affects to dopamine antagonists (oculogyric crises plus neuroleptic malignant syndrome). Investigation was unremarkable other than the presence of CSF lymphocytosis and oligoclonal bands. Despite prolonged in-patient stays and intensive care management, both patients have made complete recoveries. We believe these cases support the hypothesis that this syndrome is an inflammatory encephalitis that specifically effects dopamine neurotransmission.
DOI: 10.1002/mds.21664
PubMed: 17914719
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pubmed:17914719Le document en format XML
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<author><name sortKey="Dale, Russell C" sort="Dale, Russell C" uniqKey="Dale R" first="Russell C" last="Dale">Russell C. Dale</name>
<affiliation wicri:level="1"><nlm:affiliation>Discipline of Paediatrics and Child Health, Faculty of Medicine, University of Sydney, Sydney, New South Wales, Australia. russeld@chw.edu.au</nlm:affiliation>
<country xml:lang="fr">Australie</country>
<wicri:regionArea>Discipline of Paediatrics and Child Health, Faculty of Medicine, University of Sydney, Sydney, New South Wales</wicri:regionArea>
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<author><name sortKey="Webster, Richard" sort="Webster, Richard" uniqKey="Webster R" first="Richard" last="Webster">Richard Webster</name>
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<author><name sortKey="Gill, Deepak" sort="Gill, Deepak" uniqKey="Gill D" first="Deepak" last="Gill">Deepak Gill</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Contemporary encephalitis lethargica presenting with agitated catatonia, stereotypy, and dystonia-parkinsonism.</title>
<author><name sortKey="Dale, Russell C" sort="Dale, Russell C" uniqKey="Dale R" first="Russell C" last="Dale">Russell C. Dale</name>
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<term>Child</term>
<term>Dopamine (physiology)</term>
<term>Dystonia (diagnosis)</term>
<term>Dystonia (etiology)</term>
<term>Dystonia (therapy)</term>
<term>Humans</term>
<term>Intensive Care</term>
<term>Male</term>
<term>Parkinson Disease, Postencephalitic (complications)</term>
<term>Parkinson Disease, Postencephalitic (diagnosis)</term>
<term>Parkinson Disease, Postencephalitic (therapy)</term>
<term>Recovery of Function</term>
<term>Stereotypic Movement Disorder (diagnosis)</term>
<term>Stereotypic Movement Disorder (etiology)</term>
<term>Stereotypic Movement Disorder (therapy)</term>
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<term>Dystonia</term>
<term>Parkinson Disease, Postencephalitic</term>
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<term>Dystonia</term>
<term>Parkinson Disease, Postencephalitic</term>
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<keywords scheme="MESH" xml:lang="en"><term>Adolescent</term>
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<term>Humans</term>
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<front><div type="abstract" xml:lang="en">Encephalitis lethargica (EL) syndrome was classically described by Von Economo and has somnolent-ophthalmoplegic, hyperkinetic, and amyostatic-akinetic forms. We describe 2 recent cases of EL characterized by an acute encephalitis with mixed movement disorders (dystonia-Parkinsonism plus stereotypy) and psychiatric disorders (agitated catatonia, coprolalia, and echo phenomena). Both patients suffered concurrent hyperkinetic and Parkinsonian features resulting in therapeutic challenges. Bradykinetic features responded to dopamine replacement therapy and both patients also had adverse affects to dopamine antagonists (oculogyric crises plus neuroleptic malignant syndrome). Investigation was unremarkable other than the presence of CSF lymphocytosis and oligoclonal bands. Despite prolonged in-patient stays and intensive care management, both patients have made complete recoveries. We believe these cases support the hypothesis that this syndrome is an inflammatory encephalitis that specifically effects dopamine neurotransmission.</div>
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<Abstract><AbstractText>Encephalitis lethargica (EL) syndrome was classically described by Von Economo and has somnolent-ophthalmoplegic, hyperkinetic, and amyostatic-akinetic forms. We describe 2 recent cases of EL characterized by an acute encephalitis with mixed movement disorders (dystonia-Parkinsonism plus stereotypy) and psychiatric disorders (agitated catatonia, coprolalia, and echo phenomena). Both patients suffered concurrent hyperkinetic and Parkinsonian features resulting in therapeutic challenges. Bradykinetic features responded to dopamine replacement therapy and both patients also had adverse affects to dopamine antagonists (oculogyric crises plus neuroleptic malignant syndrome). Investigation was unremarkable other than the presence of CSF lymphocytosis and oligoclonal bands. Despite prolonged in-patient stays and intensive care management, both patients have made complete recoveries. We believe these cases support the hypothesis that this syndrome is an inflammatory encephalitis that specifically effects dopamine neurotransmission.</AbstractText>
<CopyrightInformation>(c) 2007 Movement Disorder Society.</CopyrightInformation>
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