Involvement of the medial pallidum in focal myoclonic dystonia: A clinical and neurophysiological case study.
Identifieur interne : 003B23 ( PubMed/Corpus ); précédent : 003B22; suivant : 003B24Involvement of the medial pallidum in focal myoclonic dystonia: A clinical and neurophysiological case study.
Auteurs : Xuguang Liu ; Ivan C. Griffin ; Simon G. Parkin ; R Christopher Miall ; Jeremy G. Rowe ; Ralph P. Gregory ; Richard B. Scott ; Tipu Z. Aziz ; John F. SteinSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2002.
English descriptors
- KwdEn :
- Adult, Dominance, Cerebral (physiology), Dystonic Disorders (diagnosis), Dystonic Disorders (genetics), Dystonic Disorders (physiopathology), Dystonic Disorders (therapy), Electric Stimulation Therapy, Electrodes, Implanted, Electroencephalography, Electromyography, Globus Pallidus (physiopathology), Humans, Male, Neck Muscles (innervation).
- MESH :
- diagnosis : Dystonic Disorders.
- genetics : Dystonic Disorders.
- innervation : Neck Muscles.
- physiology : Dominance, Cerebral.
- physiopathology : Dystonic Disorders, Globus Pallidus.
- therapy : Dystonic Disorders.
- Adult, Electric Stimulation Therapy, Electrodes, Implanted, Electroencephalography, Electromyography, Humans, Male.
Abstract
We successfully treated a patient with familial myoclonic dystonia (FMD), which primarily affected his neck muscles, with bilateral deep brain stimulation (DBS) to the medial pallidum, and investigated the role of the medial pallidum in FMD. A patient with FMD underwent bilateral implantation of DBS electrodes during which field potentials (FPs) in the medial pallidum and electromyograms (EMGs) from the affected neck muscles were recorded. The effects of high-frequency DBS to the medial pallidum on the FMD were also assessed by recording EMGs during and immediately after implantation, as well as 6 days and 8 weeks postoperatively. During spontaneous myoclonic episodes, increased FPs oscillating at 4 and 8 Hz were recorded from the medial pallidum; these correlated strongly with phasic EMG activity at the same frequencies in the contralateral affected muscles. The EMG activity was suppressed by stimulating the contralateral medial pallidum at 100 Hz during the operation and continuous bilateral DBS from an implanted stimulator abolished myoclonic activity even more effectively postoperatively. The phasic pallidal activity correlated with and led the myoclonic muscle activity, and the myoclonus was suppressed by bilateral pallidal DBS, suggesting that the medial pallidum was involved in the generation of the myoclonic activity. High-frequency DBS may suppress the myoclonus by desynchronising abnormal pallidal oscillations. This case study has significant clinical implications, because at present, there is no effective treatment for focal myoclonic dystonia.
PubMed: 11921122
Links to Exploration step
pubmed:11921122Le document en format XML
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<author><name sortKey="Liu, Xuguang" sort="Liu, Xuguang" uniqKey="Liu X" first="Xuguang" last="Liu">Xuguang Liu</name>
<affiliation><nlm:affiliation>University Laboratory of Physiology, Parks Road, Oxford, United Kingdom. xuguang.liu@physiol.ox.ac.uk</nlm:affiliation>
</affiliation>
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<author><name sortKey="Griffin, Ivan C" sort="Griffin, Ivan C" uniqKey="Griffin I" first="Ivan C" last="Griffin">Ivan C. Griffin</name>
</author>
<author><name sortKey="Parkin, Simon G" sort="Parkin, Simon G" uniqKey="Parkin S" first="Simon G" last="Parkin">Simon G. Parkin</name>
</author>
<author><name sortKey="Miall, R Christopher" sort="Miall, R Christopher" uniqKey="Miall R" first="R Christopher" last="Miall">R Christopher Miall</name>
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<author><name sortKey="Rowe, Jeremy G" sort="Rowe, Jeremy G" uniqKey="Rowe J" first="Jeremy G" last="Rowe">Jeremy G. Rowe</name>
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<author><name sortKey="Gregory, Ralph P" sort="Gregory, Ralph P" uniqKey="Gregory R" first="Ralph P" last="Gregory">Ralph P. Gregory</name>
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<author><name sortKey="Scott, Richard B" sort="Scott, Richard B" uniqKey="Scott R" first="Richard B" last="Scott">Richard B. Scott</name>
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<author><name sortKey="Aziz, Tipu Z" sort="Aziz, Tipu Z" uniqKey="Aziz T" first="Tipu Z" last="Aziz">Tipu Z. Aziz</name>
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<author><name sortKey="Stein, John F" sort="Stein, John F" uniqKey="Stein J" first="John F" last="Stein">John F. Stein</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Involvement of the medial pallidum in focal myoclonic dystonia: A clinical and neurophysiological case study.</title>
<author><name sortKey="Liu, Xuguang" sort="Liu, Xuguang" uniqKey="Liu X" first="Xuguang" last="Liu">Xuguang Liu</name>
<affiliation><nlm:affiliation>University Laboratory of Physiology, Parks Road, Oxford, United Kingdom. xuguang.liu@physiol.ox.ac.uk</nlm:affiliation>
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<author><name sortKey="Griffin, Ivan C" sort="Griffin, Ivan C" uniqKey="Griffin I" first="Ivan C" last="Griffin">Ivan C. Griffin</name>
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<author><name sortKey="Parkin, Simon G" sort="Parkin, Simon G" uniqKey="Parkin S" first="Simon G" last="Parkin">Simon G. Parkin</name>
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<author><name sortKey="Miall, R Christopher" sort="Miall, R Christopher" uniqKey="Miall R" first="R Christopher" last="Miall">R Christopher Miall</name>
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<author><name sortKey="Rowe, Jeremy G" sort="Rowe, Jeremy G" uniqKey="Rowe J" first="Jeremy G" last="Rowe">Jeremy G. Rowe</name>
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<author><name sortKey="Gregory, Ralph P" sort="Gregory, Ralph P" uniqKey="Gregory R" first="Ralph P" last="Gregory">Ralph P. Gregory</name>
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<author><name sortKey="Scott, Richard B" sort="Scott, Richard B" uniqKey="Scott R" first="Richard B" last="Scott">Richard B. Scott</name>
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<author><name sortKey="Aziz, Tipu Z" sort="Aziz, Tipu Z" uniqKey="Aziz T" first="Tipu Z" last="Aziz">Tipu Z. Aziz</name>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term>
<term>Dominance, Cerebral (physiology)</term>
<term>Dystonic Disorders (diagnosis)</term>
<term>Dystonic Disorders (genetics)</term>
<term>Dystonic Disorders (physiopathology)</term>
<term>Dystonic Disorders (therapy)</term>
<term>Electric Stimulation Therapy</term>
<term>Electrodes, Implanted</term>
<term>Electroencephalography</term>
<term>Electromyography</term>
<term>Globus Pallidus (physiopathology)</term>
<term>Humans</term>
<term>Male</term>
<term>Neck Muscles (innervation)</term>
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<keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Dystonic Disorders</term>
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<keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Dystonic Disorders</term>
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<keywords scheme="MESH" qualifier="innervation" xml:lang="en"><term>Neck Muscles</term>
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<keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Dominance, Cerebral</term>
</keywords>
<keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Dystonic Disorders</term>
<term>Globus Pallidus</term>
</keywords>
<keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Dystonic Disorders</term>
</keywords>
<keywords scheme="MESH" xml:lang="en"><term>Adult</term>
<term>Electric Stimulation Therapy</term>
<term>Electrodes, Implanted</term>
<term>Electroencephalography</term>
<term>Electromyography</term>
<term>Humans</term>
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<front><div type="abstract" xml:lang="en">We successfully treated a patient with familial myoclonic dystonia (FMD), which primarily affected his neck muscles, with bilateral deep brain stimulation (DBS) to the medial pallidum, and investigated the role of the medial pallidum in FMD. A patient with FMD underwent bilateral implantation of DBS electrodes during which field potentials (FPs) in the medial pallidum and electromyograms (EMGs) from the affected neck muscles were recorded. The effects of high-frequency DBS to the medial pallidum on the FMD were also assessed by recording EMGs during and immediately after implantation, as well as 6 days and 8 weeks postoperatively. During spontaneous myoclonic episodes, increased FPs oscillating at 4 and 8 Hz were recorded from the medial pallidum; these correlated strongly with phasic EMG activity at the same frequencies in the contralateral affected muscles. The EMG activity was suppressed by stimulating the contralateral medial pallidum at 100 Hz during the operation and continuous bilateral DBS from an implanted stimulator abolished myoclonic activity even more effectively postoperatively. The phasic pallidal activity correlated with and led the myoclonic muscle activity, and the myoclonus was suppressed by bilateral pallidal DBS, suggesting that the medial pallidum was involved in the generation of the myoclonic activity. High-frequency DBS may suppress the myoclonus by desynchronising abnormal pallidal oscillations. This case study has significant clinical implications, because at present, there is no effective treatment for focal myoclonic dystonia.</div>
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<Title>Movement disorders : official journal of the Movement Disorder Society</Title>
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<ArticleTitle>Involvement of the medial pallidum in focal myoclonic dystonia: A clinical and neurophysiological case study.</ArticleTitle>
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<Abstract><AbstractText>We successfully treated a patient with familial myoclonic dystonia (FMD), which primarily affected his neck muscles, with bilateral deep brain stimulation (DBS) to the medial pallidum, and investigated the role of the medial pallidum in FMD. A patient with FMD underwent bilateral implantation of DBS electrodes during which field potentials (FPs) in the medial pallidum and electromyograms (EMGs) from the affected neck muscles were recorded. The effects of high-frequency DBS to the medial pallidum on the FMD were also assessed by recording EMGs during and immediately after implantation, as well as 6 days and 8 weeks postoperatively. During spontaneous myoclonic episodes, increased FPs oscillating at 4 and 8 Hz were recorded from the medial pallidum; these correlated strongly with phasic EMG activity at the same frequencies in the contralateral affected muscles. The EMG activity was suppressed by stimulating the contralateral medial pallidum at 100 Hz during the operation and continuous bilateral DBS from an implanted stimulator abolished myoclonic activity even more effectively postoperatively. The phasic pallidal activity correlated with and led the myoclonic muscle activity, and the myoclonus was suppressed by bilateral pallidal DBS, suggesting that the medial pallidum was involved in the generation of the myoclonic activity. High-frequency DBS may suppress the myoclonus by desynchronising abnormal pallidal oscillations. This case study has significant clinical implications, because at present, there is no effective treatment for focal myoclonic dystonia.</AbstractText>
<CopyrightInformation>Copyright 2002 Movement Disorder Society.</CopyrightInformation>
</Abstract>
<AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Liu</LastName>
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<AffiliationInfo><Affiliation>University Laboratory of Physiology, Parks Road, Oxford, United Kingdom. xuguang.liu@physiol.ox.ac.uk</Affiliation>
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<MeshHeading><DescriptorName MajorTopicYN="N" UI="D008297">Male</DescriptorName>
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<MeshHeading><DescriptorName MajorTopicYN="N" UI="D009334">Neck Muscles</DescriptorName>
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