Onset of Huntington disease: Can it be purely cognitive?
Identifieur interne : 000021 ( Pmc/Checkpoint ); précédent : 000020; suivant : 000022Onset of Huntington disease: Can it be purely cognitive?
Auteurs : Jane S. Paulsen [États-Unis] ; Jeffrey D. Long [États-Unis]Source :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2014.
Abstract
Knowledge of the cognitive manifestation of Huntington disease has burgeoned over the past two decades. Many studies from independent datasets have shown cognitive impairment is evident prior to motor diagnosis and annual cognitive decline is a robust marker of disease progression. Additionally, cognition is a critical concern to patients and families and is associated with meaningful outcomes including functional capacity, driving, loss of accustomed work and quality of life. In the past few years, Huntington disease animal models of cognition have increased, preparing for preclinical experimental therapeutics with cognitive endpoints. A longitudinal analysis of cognitive variables was conducted with 559 gene-positive cases and 233 controls showing no signs of motor abnormalities over approximately a three year period. Results show there were statistically significant differences in rate of annual change for some cognitive variables, such that the cases group had worsening performance over time. These findings show cognitive deterioration can be seen in persons with the Huntington disease gene expansion with no overt motor signs or symptoms, suggesting that cognitive onset of Huntington disease may precede motor.
Url:
DOI: 10.1002/mds.25997
PubMed: 25142616
PubMed Central: 4193504
Affiliations:
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<front><div type="abstract" xml:lang="en"><p id="P1">Knowledge of the cognitive manifestation of Huntington disease has burgeoned over the past two decades. Many studies from independent datasets have shown cognitive impairment is evident prior to motor diagnosis and annual cognitive decline is a robust marker of disease progression. Additionally, cognition is a critical concern to patients and families and is associated with meaningful outcomes including functional capacity, driving, loss of accustomed work and quality of life. In the past few years, Huntington disease animal models of cognition have increased, preparing for preclinical experimental therapeutics with cognitive endpoints. A longitudinal analysis of cognitive variables was conducted with 559 gene-positive cases and 233 controls showing no signs of motor abnormalities over approximately a three year period. Results show there were statistically significant differences in rate of annual change for some cognitive variables, such that the cases group had worsening performance over time. These findings show cognitive deterioration can be seen in persons with the Huntington disease gene expansion with no overt motor signs or symptoms, suggesting that cognitive onset of Huntington disease may precede motor.</p>
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<contrib-group><contrib contrib-type="author"><name><surname>Paulsen</surname>
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<xref ref-type="aff" rid="A1">1</xref>
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<xref ref-type="aff" rid="A3">3</xref>
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Department of Psychiatry, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA</aff>
<aff id="A2"><label>2</label>
Department of Neurology, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA</aff>
<aff id="A3"><label>3</label>
Department of Psychology, The University of Iowa, Iowa City, IA, USA</aff>
<aff id="A4"><label>4</label>
Department of Biostatistics, College of Public Health, The University of Iowa, Iowa City, IA, USA</aff>
<author-notes><corresp id="cor1"><label>*</label>
<bold>Correspondence to:</bold>
Jane S. Paulsen, Carver College of Medicine, Carver Chair in Neuroscience and Professor of Neurology, Psychiatry and Psychology, 1–305 Medical Education Building, Iowa City, IA 52242-1000, USA. 319-353-4551, <email>predict-publications@uiowa.edu</email>
; <email>jane-paulsen@uiowa.edu</email>
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<pmc-comment>elocation-id from pubmed: 10.1002/mds.25997</pmc-comment>
<abstract><p id="P1">Knowledge of the cognitive manifestation of Huntington disease has burgeoned over the past two decades. Many studies from independent datasets have shown cognitive impairment is evident prior to motor diagnosis and annual cognitive decline is a robust marker of disease progression. Additionally, cognition is a critical concern to patients and families and is associated with meaningful outcomes including functional capacity, driving, loss of accustomed work and quality of life. In the past few years, Huntington disease animal models of cognition have increased, preparing for preclinical experimental therapeutics with cognitive endpoints. A longitudinal analysis of cognitive variables was conducted with 559 gene-positive cases and 233 controls showing no signs of motor abnormalities over approximately a three year period. Results show there were statistically significant differences in rate of annual change for some cognitive variables, such that the cases group had worsening performance over time. These findings show cognitive deterioration can be seen in persons with the Huntington disease gene expansion with no overt motor signs or symptoms, suggesting that cognitive onset of Huntington disease may precede motor.</p>
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