Multiple System Atrophy: Prognostic Indicators of Survival
Identifieur interne : 000510 ( Main/Exploration ); précédent : 000509; suivant : 000511Multiple System Atrophy: Prognostic Indicators of Survival
Auteurs : Juan J. Figueroa [États-Unis] ; Wolfgang Singer [États-Unis] ; Ajay Parsaik [États-Unis] ; Eduardo E. Benarroch [États-Unis] ; J. Eric Ahlskog [États-Unis] ; Robert D. Fealey [États-Unis] ; Joseph E. Parisi [États-Unis] ; Paola Sandroni [États-Unis] ; Jay Mandrekar [États-Unis] ; Valeria Iodice [Royaume-Uni] ; Phillip A. Low [États-Unis] ; James H. Bower [États-Unis]Source :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 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
Neurologic and autonomic presentation in multiple system atrophy (MSA) may predict early mortality. Quantification of early autonomic failure as mortality predictor is lacking.
Early neurologic 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;
Our data suggests that early development of severe generalized autonomic failure more than triples the risk of shorter survival in patients with MSA.
Url:
DOI: 10.1002/mds.25927
PubMed: 24909319
PubMed Central: 4139446
Affiliations:
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Le document en format XML
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<author><name sortKey="Benarroch, Eduardo E" sort="Benarroch, Eduardo E" uniqKey="Benarroch E" first="Eduardo E." last="Benarroch">Eduardo E. Benarroch</name>
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<country xml:lang="fr">États-Unis</country>
<placeName><region type="state">Minnesota</region>
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<author><name sortKey="Sandroni, Paola" sort="Sandroni, Paola" uniqKey="Sandroni P" first="Paola" last="Sandroni">Paola Sandroni</name>
<affiliation wicri:level="2"><nlm:aff id="A2">Department of Neurology, Mayo Clinic, Rochester, MN</nlm:aff>
<country xml:lang="fr">États-Unis</country>
<placeName><region type="state">Minnesota</region>
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<wicri:cityArea>Department of Neurology, Mayo Clinic, Rochester</wicri:cityArea>
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<author><name sortKey="Mandrekar, Jay" sort="Mandrekar, Jay" uniqKey="Mandrekar J" first="Jay" last="Mandrekar">Jay Mandrekar</name>
<affiliation wicri:level="2"><nlm:aff id="A5">Department of Health Sciences Research, Mayo Clinic Rochester, MN</nlm:aff>
<country xml:lang="fr">États-Unis</country>
<placeName><region type="state">Minnesota</region>
</placeName>
<wicri:cityArea>Department of Health Sciences Research, Mayo Clinic Rochester</wicri:cityArea>
</affiliation>
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<author><name sortKey="Iodice, Valeria" sort="Iodice, Valeria" uniqKey="Iodice V" first="Valeria" last="Iodice">Valeria Iodice</name>
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<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
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<author><name sortKey="Low, Phillip A" sort="Low, Phillip A" uniqKey="Low P" first="Phillip A." last="Low">Phillip A. Low</name>
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<country xml:lang="fr">États-Unis</country>
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</placeName>
<wicri:cityArea>Department of Neurology, Mayo Clinic, Rochester</wicri:cityArea>
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<author><name sortKey="Bower, James H" sort="Bower, James H" uniqKey="Bower J" first="James H." last="Bower">James H. Bower</name>
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<country xml:lang="fr">États-Unis</country>
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</placeName>
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<series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title>
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<idno type="eISSN">1531-8257</idno>
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<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>
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<front><div type="abstract" xml:lang="en"><sec id="S1"><title>Background</title>
<p id="P1">Neurologic and autonomic presentation in multiple system atrophy (MSA) may predict early mortality. Quantification of early autonomic failure as mortality predictor is lacking.</p>
</sec>
<sec id="S2"><title>Methods</title>
<p id="P2">Early neurologic 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.</p>
</sec>
<sec id="S3"><title>Results</title>
<p id="P3">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; <italic>P</italic>
=0.036), and early requirement of bladder catheterization (7.3 [3.1–10.2] vs. 13.7 [8.5–14.9] years; <italic>P</italic>
=0.003) compared to 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]; <italic>P</italic>
=0.03), early requirement of bladder catheterization (7.9 [1.88–38.63]; <italic>P</italic>
=0.004) and early generalized (composite autonomic severity score ≥ 6) autonomic failure (2.8 [1.01–9.26]; <italic>P</italic>
=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.</p>
</sec>
<sec id="S4"><title>Conclusions</title>
<p id="P4">Our data suggests that early development of severe generalized autonomic failure more than triples the risk of shorter survival in patients with MSA.</p>
</sec>
</div>
</front>
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<name sortKey="Parsaik, Ajay" sort="Parsaik, Ajay" uniqKey="Parsaik A" first="Ajay" last="Parsaik">Ajay Parsaik</name>
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<country name="Royaume-Uni"><region name="Angleterre"><name sortKey="Iodice, Valeria" sort="Iodice, Valeria" uniqKey="Iodice V" first="Valeria" last="Iodice">Valeria Iodice</name>
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