Minimal change multiple system atrophy: an aggressive variant?
Identifieur interne : 000171 ( PubMed/Checkpoint ); précédent : 000170; suivant : 000172Minimal 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.
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
Affiliations:
- Canada, Japon, Royaume-Uni
- Angleterre, Grand Londres, Ontario
- Londres, Toronto
- Université de Toronto
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Holton</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2015">2015</date><idno type="RBID">pubmed:25854893</idno><idno type="pmid">25854893</idno><idno type="doi">10.1002/mds.26220</idno><idno type="wicri:Area/PubMed/Corpus">000197</idno><idno type="wicri:Area/PubMed/Curation">000197</idno><idno type="wicri:Area/PubMed/Checkpoint">000171</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Minimal change multiple system atrophy: an aggressive variant?</title><author><name sortKey="Ling, Helen" sort="Ling, Helen" uniqKey="Ling H" first="Helen" last="Ling">Helen Ling</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Asi, Yasmine T" sort="Asi, Yasmine T" uniqKey="Asi Y" first="Yasmine T" last="Asi">Yasmine T. Asi</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Petrovic, Igor N" sort="Petrovic, Igor N" uniqKey="Petrovic I" first="Igor N" last="Petrovic">Igor N. Petrovic</name><affiliation wicri:level="3"><nlm:affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London</wicri:regionArea><placeName><settlement type="city">Londres</settlement><region type="country">Angleterre</region><region type="région" nuts="1">Grand Londres</region></placeName></affiliation></author><author><name sortKey="Ahmed, Zeshan" sort="Ahmed, Zeshan" uniqKey="Ahmed Z" first="Zeshan" last="Ahmed">Zeshan Ahmed</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Prashanth, L K" sort="Prashanth, L K" uniqKey="Prashanth L" first="L K" last="Prashanth">L K Prashanth</name><affiliation wicri:level="4"><nlm:affiliation>University of Toronto, Toronto, Canada.</nlm:affiliation><country xml:lang="fr">Canada</country><wicri:regionArea>University of Toronto, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation></author><author><name sortKey="Hazrati, Lili Naz" sort="Hazrati, Lili Naz" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name><affiliation wicri:level="4"><nlm:affiliation>University of Toronto, Toronto, Canada.</nlm:affiliation><country xml:lang="fr">Canada</country><wicri:regionArea>University of Toronto, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation></author><author><name sortKey="Nishizawa, Masatoyo" sort="Nishizawa, Masatoyo" uniqKey="Nishizawa M" first="Masatoyo" last="Nishizawa">Masatoyo Nishizawa</name><affiliation wicri:level="1"><nlm:affiliation>Department of Neurology, Niigata University, Japan.</nlm:affiliation><country xml:lang="fr">Japon</country><wicri:regionArea>Department of Neurology, Niigata University</wicri:regionArea><wicri:noRegion>Niigata University</wicri:noRegion></affiliation></author><author><name sortKey="Ozawa, Tetsutaro" sort="Ozawa, Tetsutaro" uniqKey="Ozawa T" first="Tetsutaro" last="Ozawa">Tetsutaro Ozawa</name><affiliation wicri:level="1"><nlm:affiliation>Department of Neurology, Niigata University, Japan.</nlm:affiliation><country xml:lang="fr">Japon</country><wicri:regionArea>Department of Neurology, Niigata University</wicri:regionArea><wicri:noRegion>Niigata University</wicri:noRegion></affiliation></author><author><name sortKey="Lang, Anthony" sort="Lang, Anthony" uniqKey="Lang A" first="Anthony" last="Lang">Anthony Lang</name><affiliation wicri:level="4"><nlm:affiliation>University of Toronto, Toronto, Canada.</nlm:affiliation><country xml:lang="fr">Canada</country><wicri:regionArea>University of Toronto, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation></author><author><name sortKey="Lees, Andrew J" sort="Lees, Andrew J" uniqKey="Lees A" first="Andrew J" last="Lees">Andrew J. Lees</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Revesz, Tamas" sort="Revesz, Tamas" uniqKey="Revesz T" first="Tamas" last="Revesz">Tamas Revesz</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Holton, Janice L" sort="Holton, Janice L" uniqKey="Holton J" first="Janice L" last="Holton">Janice L. Holton</name><affiliation wicri:level="1"><nlm:affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology</wicri:regionArea><wicri:noRegion>UCL Institute of Neurology</wicri:noRegion></affiliation></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="eISSN">1531-8257</idno><imprint><date when="2015" type="published">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><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></TEI><pubmed><MedlineCitation Owner="NLM" Status="In-Process"><PMID Version="1">25854893</PMID><DateCreated><Year>2015</Year><Month>06</Month><Day>23</Day></DateCreated><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><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Ling</LastName><ForeName>Helen</ForeName><Initials>H</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Asi</LastName><ForeName>Yasmine T</ForeName><Initials>YT</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Petrovic</LastName><ForeName>Igor N</ForeName><Initials>IN</Initials><AffiliationInfo><Affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Institute of Neurology, School of Medicine, Belgrade, Serbia.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Ahmed</LastName><ForeName>Zeshan</ForeName><Initials>Z</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Prashanth</LastName><ForeName>L K</ForeName><Initials>LK</Initials><AffiliationInfo><Affiliation>University of Toronto, Toronto, Canada.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Edmond J. Safra Program in Parkinson's Disease, Toronto Western Hospital, Toronto, Canada.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Hazrati</LastName><ForeName>Lili-Naz</ForeName><Initials>LN</Initials><AffiliationInfo><Affiliation>University of Toronto, Toronto, Canada.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Edmond J. Safra Program in Parkinson's Disease, Toronto Western Hospital, Toronto, Canada.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Nishizawa</LastName><ForeName>Masatoyo</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>Department of Neurology, Niigata University, Japan.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Ozawa</LastName><ForeName>Tetsutaro</ForeName><Initials>T</Initials><AffiliationInfo><Affiliation>Department of Neurology, Niigata University, Japan.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lang</LastName><ForeName>Anthony</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>University of Toronto, Toronto, Canada.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Edmond J. Safra Program in Parkinson's Disease, Toronto Western Hospital, Toronto, Canada.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lees</LastName><ForeName>Andrew J</ForeName><Initials>AJ</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Reta Lila Weston Institute of Neurological Studies, UCL Institute of Neurology, London, UK.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Revesz</LastName><ForeName>Tamas</ForeName><Initials>T</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Holton</LastName><ForeName>Janice L</ForeName><Initials>JL</Initials><AffiliationInfo><Affiliation>Queen Square Brain Bank, Department of Molecular Neuroscience, UCL Institute of Neurology, UK.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D013485">Research Support, Non-U.S. Gov't</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2015</Year><Month>04</Month><Day>08</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov Disord</MedlineTA><NlmUniqueID>8610688</NlmUniqueID><ISSNLinking>0885-3185</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><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></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2014</Year><Month>8</Month><Day>8</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2015</Year><Month>2</Month><Day>22</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2015</Year><Month>3</Month><Day>2</Day></PubMedPubDate><PubMedPubDate PubStatus="aheadofprint"><Year>2015</Year><Month>4</Month><Day>8</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2015</Year><Month>4</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2015</Year><Month>4</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2015</Year><Month>4</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">25854893</ArticleId><ArticleId IdType="doi">10.1002/mds.26220</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>Canada</li><li>Japon</li><li>Royaume-Uni</li></country><region><li>Angleterre</li><li>Grand Londres</li><li>Ontario</li></region><settlement><li>Londres</li><li>Toronto</li></settlement><orgName><li>Université de Toronto</li></orgName></list><tree><country name="Royaume-Uni"><noRegion><name sortKey="Ling, Helen" sort="Ling, Helen" uniqKey="Ling H" first="Helen" last="Ling">Helen Ling</name></noRegion><name sortKey="Ahmed, Zeshan" sort="Ahmed, Zeshan" uniqKey="Ahmed Z" first="Zeshan" last="Ahmed">Zeshan Ahmed</name><name sortKey="Asi, Yasmine T" sort="Asi, Yasmine T" uniqKey="Asi Y" first="Yasmine T" last="Asi">Yasmine T. Asi</name><name sortKey="Holton, Janice L" sort="Holton, Janice L" uniqKey="Holton J" first="Janice L" last="Holton">Janice L. Holton</name><name sortKey="Lees, Andrew J" sort="Lees, Andrew J" uniqKey="Lees A" first="Andrew J" last="Lees">Andrew J. Lees</name><name sortKey="Petrovic, Igor N" sort="Petrovic, Igor N" uniqKey="Petrovic I" first="Igor N" last="Petrovic">Igor N. 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