Minimal change multiple system atrophy: an aggressive variant?
Identifieur interne : 000489 ( PubMed/Curation ); précédent : 000488; suivant : 000490Minimal change multiple system atrophy: an aggressive variant?
Auteurs : Helen Ling [Royaume-Uni] ; Yasmine T. Asi [Royaume-Uni] ; Igor N. Petrovic [Royaume-Uni] ; Zeshan Ahmed [Royaume-Uni] ; L K Prashanth [Canada] ; Lili-Naz Hazrati [Canada] ; Masatoyo Nishizawa [Japon] ; Tetsutaro Ozawa [Japon] ; Anthony Lang [Canada] ; Andrew J. Lees [Royaume-Uni] ; Tamas Revesz [Royaume-Uni] ; Janice L. Holton [Royaume-Uni]Source :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2015.
English descriptors
- KwdEn :
- Adult, Age of Onset, Aged, Aged, 80 and over, Brain (metabolism), Brain (pathology), Humans, Inclusion Bodies (metabolism), Inclusion Bodies (pathology), Male, Middle Aged, Multiple System Atrophy (classification), Multiple System Atrophy (metabolism), Multiple System Atrophy (pathology), Tissue Banks, alpha-Synuclein (metabolism).
- MESH :
- chemical , metabolism : alpha-Synuclein.
- classification : Multiple System Atrophy.
- metabolism : Brain, Inclusion Bodies, Multiple System Atrophy.
- pathology : Brain, Inclusion Bodies, Multiple System Atrophy.
- Adult, Age of Onset, Aged, Aged, 80 and over, Humans, Male, Middle Aged, Tissue Banks.
Abstract
Glial cytoplasmic inclusions containing α-synuclein are the pathological hallmark of multiple system atrophy (MSA). Minimal change (MC-MSA) is an unusual MSA subtype with neuronal loss largely restricted to the substantia nigra and locus coeruleus.
DOI: 10.1002/mds.26220
PubMed: 25854893
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<term>Humans</term>
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<term>Inclusion Bodies (pathology)</term>
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<front><div type="abstract" xml:lang="en">Glial cytoplasmic inclusions containing α-synuclein are the pathological hallmark of multiple system atrophy (MSA). Minimal change (MC-MSA) is an unusual MSA subtype with neuronal loss largely restricted to the substantia nigra and locus coeruleus.</div>
</front>
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<DateCreated><Year>2015</Year>
<Month>06</Month>
<Day>23</Day>
</DateCreated>
<DateCompleted><Year>2016</Year>
<Month>04</Month>
<Day>04</Day>
</DateCompleted>
<DateRevised><Year>2015</Year>
<Month>06</Month>
<Day>23</Day>
</DateRevised>
<Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1531-8257</ISSN>
<JournalIssue CitedMedium="Internet"><Volume>30</Volume>
<Issue>7</Issue>
<PubDate><Year>2015</Year>
<Month>Jun</Month>
</PubDate>
</JournalIssue>
<Title>Movement disorders : official journal of the Movement Disorder Society</Title>
<ISOAbbreviation>Mov. Disord.</ISOAbbreviation>
</Journal>
<ArticleTitle>Minimal change multiple system atrophy: an aggressive variant?</ArticleTitle>
<Pagination><MedlinePgn>960-7</MedlinePgn>
</Pagination>
<ELocationID EIdType="doi" ValidYN="Y">10.1002/mds.26220</ELocationID>
<Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Glial cytoplasmic inclusions containing α-synuclein are the pathological hallmark of multiple system atrophy (MSA). Minimal change (MC-MSA) is an unusual MSA subtype with neuronal loss largely restricted to the substantia nigra and locus coeruleus.</AbstractText>
<AbstractText Label="METHODS" NlmCategory="METHODS">Immunohistochemistry on selected brain regions and semiquantitative assessment were performed on six MC-MSA and eight MSA control cases.</AbstractText>
<AbstractText Label="RESULTS" NlmCategory="RESULTS">More neuronal cytoplasmic inclusions were seen in the caudate and substantia nigra in MC-MSA than in MSA controls (P = 0.002), without any statistical difference in glial cytoplasmic inclusion load in any region. Severe glial cytoplasmic inclusion load was found in the ventrolateral medulla (P = 1.0) and nucleus raphe obscurus (P = 0.4) in both groups. When compared with MSA controls, the three MC-MSA cases who had died of sudden unexpected death had an earlier age of onset (mean: 38 vs. 57.6 y, P = 0.02), a numerically shorter disease duration (mean: 5.3 vs. 8 y, P = 0.2) and a more rapid clinical progression with most of the clinical milestones reached within 3 y of presentation, suggesting an aggressive variant of MSA. Another three MC-MSA cases, who had died of unrelated concurrent diseases, had an age of onset (mean: 57.7 y) and temporal course similar to controls, had less severe neuronal loss and gliosis in the medial and dorsolateral substantia nigra subregions (P < 0.05) than in MSA controls, and could be considered as a unique group with interrupted pathological progression. Significant respiratory dysfunction and early orthostatic hypotension were observed in all MC-MSA cases.</AbstractText>
<AbstractText Label="CONCLUSIONS" NlmCategory="CONCLUSIONS">Our findings could suggest that α-synuclein-associated oligodendroglial pathology may lead to neuronal dysfunction sufficient to cause clinical symptoms before overt neuronal loss in MSA. © 2015 International Parkinson and Movement Disorder Society.</AbstractText>
<CopyrightInformation>© 2015 International Parkinson and Movement Disorder Society.</CopyrightInformation>
</Abstract>
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<ForeName>Helen</ForeName>
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</AffiliationInfo>
<AffiliationInfo><Affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</Affiliation>
</AffiliationInfo>
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<AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation>
</AffiliationInfo>
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</AffiliationInfo>
<AffiliationInfo><Affiliation>Institute of Neurology, School of Medicine, Belgrade, Serbia.</Affiliation>
</AffiliationInfo>
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<ForeName>Zeshan</ForeName>
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</AffiliationInfo>
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<Author ValidYN="Y"><LastName>Prashanth</LastName>
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</AffiliationInfo>
<AffiliationInfo><Affiliation>Edmond J. Safra Program in Parkinson's Disease, Toronto Western Hospital, Toronto, Canada.</Affiliation>
</AffiliationInfo>
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</AffiliationInfo>
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</AffiliationInfo>
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<AffiliationInfo><Affiliation>Edmond J. Safra Program in Parkinson's Disease, Toronto Western Hospital, Toronto, Canada.</Affiliation>
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</AffiliationInfo>
<AffiliationInfo><Affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</Affiliation>
</AffiliationInfo>
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</MeshHeading>
</MeshHeadingList>
<KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">Minimal change</Keyword>
<Keyword MajorTopicYN="N">alpha-synuclein</Keyword>
<Keyword MajorTopicYN="N">multiple system atrophy</Keyword>
<Keyword MajorTopicYN="N">sudden unexpected death</Keyword>
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<PubmedData><History><PubMedPubDate PubStatus="received"><Year>2014</Year>
<Month>08</Month>
<Day>08</Day>
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<PubMedPubDate PubStatus="revised"><Year>2015</Year>
<Month>02</Month>
<Day>22</Day>
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<PubMedPubDate PubStatus="accepted"><Year>2015</Year>
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<PubMedPubDate PubStatus="medline"><Year>2016</Year>
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<ArticleIdList><ArticleId IdType="pubmed">25854893</ArticleId>
<ArticleId IdType="doi">10.1002/mds.26220</ArticleId>
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