Multiple system atrophy: prognostic indicators of survival.
Identifieur interne : 000516 ( PubMed/Checkpoint ); précédent : 000515; suivant : 000517Multiple system atrophy: prognostic indicators of survival.
Auteurs : Juan J. Figueroa [États-Unis] ; Wolfgang Singer ; Ajay Parsaik ; Eduardo E. Benarroch ; J Eric Ahlskog ; Robert D. Fealey ; Joseph E. Parisi ; Paola Sandroni ; Jay Mandrekar ; Valeria Iodice ; Phillip A. Low ; James H. BowerSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2014.
English descriptors
- KwdEn :
- MESH :
- complications : Multiple System Atrophy.
- diagnosis : Multiple System Atrophy.
- etiology : Autonomic Nervous System Diseases.
- mortality : Multiple System Atrophy.
- Age of Onset, Aged, Disability Evaluation, Female, Humans, Male, Middle Aged, Prognosis, Proportional Hazards Models, Risk Factors.
Abstract
Neurological and autonomic presentation in multiple system atrophy (MSA) may predict early mortality. Quantification of early autonomic failure as a mortality predictor is lacking. Early neurological and autonomic clinical features were retrospectively reviewed in 49 MSA cases (median age at onset, 56.1 years; 16 women) confirmed by autopsy at Mayo Clinic. When available, the 10-point composite autonomic severity score derived from the autonomic reflex screen provided quantification of the degree of autonomic failure and thermoregulatory sweat test quantitated body surface anhidrosis. Symptoms at onset were autonomic in 50%, parkinsonian in 30%, and cerebellar in 20% of cases. Survival (median [95% confidence interval]) was 8.6 [6.7-10.2] years. Survival was shorter in patients with early laboratory evidence of generalized (composite autonomic severity score ≥ 6) autonomic failure (7.0 [3.9-9.8] vs. 9.8 [4.6-13.8] years; P = 0.036), and early requirement of bladder catheterization (7.3 [3.1-10.2] vs. 13.7 [8.5-14.9] years; P = 0.003) compared with those without these clinical features. On Cox proportional analysis, prognostic indicators of shorter survival were older age at onset (hazard ratio [95% confidence interval], 1.04 [1.01-1.08]; P = 0.03), early requirement of bladder catheterization (7.9 [1.88-38.63]; P = 0.004), and early generalized (composite autonomic severity score ≥ 6) autonomic failure (2.8 [1.01-9.26]; P = 0.047). Gender, phenotype, and early development of gait instability, aid-requiring ambulation, orthostatic symptoms, neurogenic bladder, or significant anhidrosis (thermoregulatory sweat test ≥ 40%) were not indicators of shorter survival. Our data suggest that early development of severe generalized autonomic failure more than triples the risk of shorter survival in patients with MSA.
DOI: 10.1002/mds.25927
PubMed: 24909319
Affiliations:
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Figueroa</name><affiliation wicri:level="2"><nlm:affiliation>Department of Neurology, Medical College of Wisconsin, Milwaukee, WI.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Wisconsin</region></placeName><wicri:cityArea>Department of Neurology, Medical College of Wisconsin, Milwaukee</wicri:cityArea></affiliation></author><author><name sortKey="Singer, Wolfgang" sort="Singer, Wolfgang" uniqKey="Singer W" first="Wolfgang" last="Singer">Wolfgang Singer</name></author><author><name sortKey="Parsaik, Ajay" sort="Parsaik, Ajay" uniqKey="Parsaik A" first="Ajay" last="Parsaik">Ajay Parsaik</name></author><author><name sortKey="Benarroch, Eduardo E" sort="Benarroch, Eduardo E" uniqKey="Benarroch E" first="Eduardo E" last="Benarroch">Eduardo E. 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Figueroa</name><affiliation wicri:level="2"><nlm:affiliation>Department of Neurology, Medical College of Wisconsin, Milwaukee, WI.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Wisconsin</region></placeName><wicri:cityArea>Department of Neurology, Medical College of Wisconsin, Milwaukee</wicri:cityArea></affiliation></author><author><name sortKey="Singer, Wolfgang" sort="Singer, Wolfgang" uniqKey="Singer W" first="Wolfgang" last="Singer">Wolfgang Singer</name></author><author><name sortKey="Parsaik, Ajay" sort="Parsaik, Ajay" uniqKey="Parsaik A" first="Ajay" last="Parsaik">Ajay Parsaik</name></author><author><name sortKey="Benarroch, Eduardo E" sort="Benarroch, Eduardo E" uniqKey="Benarroch E" first="Eduardo E" last="Benarroch">Eduardo E. Benarroch</name></author><author><name sortKey="Ahlskog, J Eric" sort="Ahlskog, J Eric" uniqKey="Ahlskog J" first="J Eric" last="Ahlskog">J Eric Ahlskog</name></author><author><name sortKey="Fealey, Robert D" sort="Fealey, Robert D" uniqKey="Fealey R" first="Robert D" last="Fealey">Robert D. Fealey</name></author><author><name sortKey="Parisi, Joseph E" sort="Parisi, Joseph E" uniqKey="Parisi J" first="Joseph E" last="Parisi">Joseph E. Parisi</name></author><author><name sortKey="Sandroni, Paola" sort="Sandroni, Paola" uniqKey="Sandroni P" first="Paola" last="Sandroni">Paola Sandroni</name></author><author><name sortKey="Mandrekar, Jay" sort="Mandrekar, Jay" uniqKey="Mandrekar J" first="Jay" last="Mandrekar">Jay Mandrekar</name></author><author><name sortKey="Iodice, Valeria" sort="Iodice, Valeria" uniqKey="Iodice V" first="Valeria" last="Iodice">Valeria Iodice</name></author><author><name sortKey="Low, Phillip A" sort="Low, Phillip A" uniqKey="Low P" first="Phillip A" last="Low">Phillip A. Low</name></author><author><name sortKey="Bower, James H" sort="Bower, James H" uniqKey="Bower J" first="James H" last="Bower">James H. Bower</name></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="2014" type="published">2014</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Age of Onset</term><term>Aged</term><term>Autonomic Nervous System Diseases (etiology)</term><term>Disability Evaluation</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Multiple System Atrophy (complications)</term><term>Multiple System Atrophy (diagnosis)</term><term>Multiple System Atrophy (mortality)</term><term>Prognosis</term><term>Proportional Hazards Models</term><term>Risk Factors</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Multiple System Atrophy</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Multiple System Atrophy</term></keywords><keywords scheme="MESH" qualifier="etiology" xml:lang="en"><term>Autonomic Nervous System Diseases</term></keywords><keywords scheme="MESH" qualifier="mortality" xml:lang="en"><term>Multiple System Atrophy</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Age of Onset</term><term>Aged</term><term>Disability Evaluation</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Prognosis</term><term>Proportional Hazards Models</term><term>Risk Factors</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Neurological and autonomic presentation in multiple system atrophy (MSA) may predict early mortality. Quantification of early autonomic failure as a mortality predictor is lacking. Early neurological and autonomic clinical features were retrospectively reviewed in 49 MSA cases (median age at onset, 56.1 years; 16 women) confirmed by autopsy at Mayo Clinic. When available, the 10-point composite autonomic severity score derived from the autonomic reflex screen provided quantification of the degree of autonomic failure and thermoregulatory sweat test quantitated body surface anhidrosis. Symptoms at onset were autonomic in 50%, parkinsonian in 30%, and cerebellar in 20% of cases. Survival (median [95% confidence interval]) was 8.6 [6.7-10.2] years. Survival was shorter in patients with early laboratory evidence of generalized (composite autonomic severity score ≥ 6) autonomic failure (7.0 [3.9-9.8] vs. 9.8 [4.6-13.8] years; P = 0.036), and early requirement of bladder catheterization (7.3 [3.1-10.2] vs. 13.7 [8.5-14.9] years; P = 0.003) compared with those without these clinical features. On Cox proportional analysis, prognostic indicators of shorter survival were older age at onset (hazard ratio [95% confidence interval], 1.04 [1.01-1.08]; P = 0.03), early requirement of bladder catheterization (7.9 [1.88-38.63]; P = 0.004), and early generalized (composite autonomic severity score ≥ 6) autonomic failure (2.8 [1.01-9.26]; P = 0.047). Gender, phenotype, and early development of gait instability, aid-requiring ambulation, orthostatic symptoms, neurogenic bladder, or significant anhidrosis (thermoregulatory sweat test ≥ 40%) were not indicators of shorter survival. Our data suggest that early development of severe generalized autonomic failure more than triples the risk of shorter survival in patients with MSA.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">24909319</PMID><DateCreated><Year>2014</Year><Month>08</Month><Day>20</Day></DateCreated><DateCompleted><Year>2015</Year><Month>04</Month><Day>20</Day></DateCompleted><DateRevised><Year>2015</Year><Month>08</Month><Day>01</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1531-8257</ISSN><JournalIssue CitedMedium="Internet"><Volume>29</Volume><Issue>9</Issue><PubDate><Year>2014</Year><Month>Aug</Month></PubDate></JournalIssue><Title>Movement disorders : official journal of the Movement Disorder Society</Title><ISOAbbreviation>Mov. Disord.</ISOAbbreviation></Journal><ArticleTitle>Multiple system atrophy: prognostic indicators of survival.</ArticleTitle><Pagination><MedlinePgn>1151-7</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1002/mds.25927</ELocationID><Abstract><AbstractText>Neurological and autonomic presentation in multiple system atrophy (MSA) may predict early mortality. Quantification of early autonomic failure as a mortality predictor is lacking. Early neurological and autonomic clinical features were retrospectively reviewed in 49 MSA cases (median age at onset, 56.1 years; 16 women) confirmed by autopsy at Mayo Clinic. When available, the 10-point composite autonomic severity score derived from the autonomic reflex screen provided quantification of the degree of autonomic failure and thermoregulatory sweat test quantitated body surface anhidrosis. Symptoms at onset were autonomic in 50%, parkinsonian in 30%, and cerebellar in 20% of cases. Survival (median [95% confidence interval]) was 8.6 [6.7-10.2] years. Survival was shorter in patients with early laboratory evidence of generalized (composite autonomic severity score ≥ 6) autonomic failure (7.0 [3.9-9.8] vs. 9.8 [4.6-13.8] years; P = 0.036), and early requirement of bladder catheterization (7.3 [3.1-10.2] vs. 13.7 [8.5-14.9] years; P = 0.003) compared with those without these clinical features. On Cox proportional analysis, prognostic indicators of shorter survival were older age at onset (hazard ratio [95% confidence interval], 1.04 [1.01-1.08]; P = 0.03), early requirement of bladder catheterization (7.9 [1.88-38.63]; P = 0.004), and early generalized (composite autonomic severity score ≥ 6) autonomic failure (2.8 [1.01-9.26]; P = 0.047). Gender, phenotype, and early development of gait instability, aid-requiring ambulation, orthostatic symptoms, neurogenic bladder, or significant anhidrosis (thermoregulatory sweat test ≥ 40%) were not indicators of shorter survival. Our data suggest that early development of severe generalized autonomic failure more than triples the risk of shorter survival in patients with MSA.</AbstractText><CopyrightInformation>© 2014 International Parkinson and Movement Disorder Society.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Figueroa</LastName><ForeName>Juan J</ForeName><Initials>JJ</Initials><AffiliationInfo><Affiliation>Department of Neurology, Medical College of Wisconsin, Milwaukee, WI.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Singer</LastName><ForeName>Wolfgang</ForeName><Initials>W</Initials></Author><Author ValidYN="Y"><LastName>Parsaik</LastName><ForeName>Ajay</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>Benarroch</LastName><ForeName>Eduardo E</ForeName><Initials>EE</Initials></Author><Author ValidYN="Y"><LastName>Ahlskog</LastName><ForeName>J Eric</ForeName><Initials>JE</Initials></Author><Author ValidYN="Y"><LastName>Fealey</LastName><ForeName>Robert D</ForeName><Initials>RD</Initials></Author><Author ValidYN="Y"><LastName>Parisi</LastName><ForeName>Joseph E</ForeName><Initials>JE</Initials></Author><Author ValidYN="Y"><LastName>Sandroni</LastName><ForeName>Paola</ForeName><Initials>P</Initials></Author><Author ValidYN="Y"><LastName>Mandrekar</LastName><ForeName>Jay</ForeName><Initials>J</Initials></Author><Author ValidYN="Y"><LastName>Iodice</LastName><ForeName>Valeria</ForeName><Initials>V</Initials></Author><Author ValidYN="Y"><LastName>Low</LastName><ForeName>Phillip A</ForeName><Initials>PA</Initials></Author><Author ValidYN="Y"><LastName>Bower</LastName><ForeName>James H</ForeName><Initials>JH</Initials></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>K23 NS075141</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>K23NS075141</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>NS 32352</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>NS 44233</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>P01 NS044233</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>P50 NS032352</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>T32 HD007447</GrantID><Acronym>HD</Acronym><Agency>NICHD NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>T32 HD07447</GrantID><Acronym>HD</Acronym><Agency>NICHD NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>U54 NS065736</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>U54 NS065736</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>UL1 TR000135</GrantID><Acronym>TR</Acronym><Agency>NCATS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>UL1 TR000135</GrantID><Acronym>TR</Acronym><Agency>NCATS NIH HHS</Agency><Country>United States</Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D052061">Research Support, N.I.H., Extramural</PublicationType><PublicationType UI="D013485">Research Support, Non-U.S. Gov't</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2014</Year><Month>06</Month><Day>07</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov 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UI="D006801">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D008297">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D008875">Middle Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D019578">Multiple System Atrophy</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000175">diagnosis</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000401">mortality</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D011379">Prognosis</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D016016">Proportional Hazards Models</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D012307">Risk Factors</DescriptorName></MeshHeading></MeshHeadingList><OtherID Source="NLM">NIHMS597796</OtherID><OtherID Source="NLM">PMC4139446</OtherID><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">MSA</Keyword><Keyword MajorTopicYN="N">autonomic failure</Keyword><Keyword MajorTopicYN="N">mortality</Keyword><Keyword MajorTopicYN="N">survival</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2014</Year><Month>2</Month><Day>5</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2014</Year><Month>4</Month><Day>24</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2014</Year><Month>5</Month><Day>4</Day></PubMedPubDate><PubMedPubDate PubStatus="aheadofprint"><Year>2014</Year><Month>6</Month><Day>7</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2014</Year><Month>6</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2014</Year><Month>6</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2015</Year><Month>4</Month><Day>22</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">24909319</ArticleId><ArticleId IdType="doi">10.1002/mds.25927</ArticleId><ArticleId IdType="pmc">PMC4139446</ArticleId><ArticleId IdType="mid">NIHMS597796</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>États-Unis</li></country><region><li>Wisconsin</li></region></list><tree><noCountry><name sortKey="Ahlskog, J Eric" sort="Ahlskog, J Eric" uniqKey="Ahlskog J" first="J Eric" last="Ahlskog">J Eric Ahlskog</name><name sortKey="Benarroch, Eduardo E" sort="Benarroch, Eduardo E" uniqKey="Benarroch E" first="Eduardo E" last="Benarroch">Eduardo E. Benarroch</name><name sortKey="Bower, James H" sort="Bower, James H" uniqKey="Bower J" first="James H" last="Bower">James H. Bower</name><name sortKey="Fealey, Robert D" sort="Fealey, Robert D" uniqKey="Fealey R" first="Robert D" last="Fealey">Robert D. Fealey</name><name sortKey="Iodice, Valeria" sort="Iodice, Valeria" uniqKey="Iodice V" first="Valeria" last="Iodice">Valeria Iodice</name><name sortKey="Low, Phillip A" sort="Low, Phillip A" uniqKey="Low P" first="Phillip A" last="Low">Phillip A. Low</name><name sortKey="Mandrekar, Jay" sort="Mandrekar, Jay" uniqKey="Mandrekar J" first="Jay" last="Mandrekar">Jay Mandrekar</name><name sortKey="Parisi, Joseph E" sort="Parisi, Joseph E" uniqKey="Parisi J" first="Joseph E" last="Parisi">Joseph E. Parisi</name><name sortKey="Parsaik, Ajay" sort="Parsaik, Ajay" uniqKey="Parsaik A" first="Ajay" last="Parsaik">Ajay Parsaik</name><name sortKey="Sandroni, Paola" sort="Sandroni, Paola" uniqKey="Sandroni P" first="Paola" last="Sandroni">Paola Sandroni</name><name sortKey="Singer, Wolfgang" sort="Singer, Wolfgang" uniqKey="Singer W" first="Wolfgang" last="Singer">Wolfgang Singer</name></noCountry><country name="États-Unis"><region name="Wisconsin"><name sortKey="Figueroa, Juan J" sort="Figueroa, Juan J" uniqKey="Figueroa J" first="Juan J" last="Figueroa">Juan J. Figueroa</name></region></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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