A retrospective study of the impact of lifestyle on age at onset of Huntington disease.
Identifieur interne : 002C75 ( Ncbi/Checkpoint ); précédent : 002C74; suivant : 002C76A retrospective study of the impact of lifestyle on age at onset of Huntington disease.
Auteurs : M Kaye Trembath [Australie] ; Zoë A. Horton ; Lynette Tippett ; Virginia Hogg ; Veronica R. Collins ; Andrew Churchyard ; Dennis Velakoulis ; Richard Roxburgh ; Martin B. DelatyckiSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2010.
Descripteurs français
- Wicri :
- geographic : Nouvelle-Zélande.
English descriptors
- KwdEn :
- Adult, Age of Onset, Aged, Cohort Studies, Female, Humans, Huntington Disease (epidemiology), Huntington Disease (genetics), Huntington Disease (psychology), Life Style, Linear Models, Male, Middle Aged, Nerve Tissue Proteins (genetics), New Zealand (epidemiology), Nuclear Proteins (genetics), Retrospective Studies, Statistics as Topic, Trinucleotide Repeat Expansion (genetics), Young Adult.
- MESH :
- chemical , genetics : Nerve Tissue Proteins, Nuclear Proteins.
- geographic , epidemiology : New Zealand.
- epidemiology : Huntington Disease.
- genetics : Huntington Disease, Trinucleotide Repeat Expansion.
- psychology : Huntington Disease.
- Adult, Age of Onset, Aged, Cohort Studies, Female, Humans, Life Style, Linear Models, Male, Middle Aged, Retrospective Studies, Statistics as Topic, Young Adult.
Abstract
In transgenic mouse models of Huntington disease (HD) environmental enrichment significantly delays disease onset. A questionnaire-based survey of 154 adults with diagnosed HD (mean 4.2 years postdiagnosis) and a known IT15 CAG repeat length, explored whether premorbid lifestyle may relate to age-at-onset (AO). Participants were drawn from HD outpatient clinics in Australia and New Zealand. Premorbid physical, intellectual, and passive activity levels were used to generate scores in the categories of leisure, nonleisure (education, occupation and domestic duties) and total lifestyle. AO was associated with increased CAG repeat length as expected (r = -0.72, P < 0.001), but also with a lifestyle that included higher levels of passive activity (r = -0.38, P < 0.001). Multiple linear regression modeling showed lifestyle passivity to be a variable independent of CAG repeat length in predicting AO (R(2) = 0.54, b = -0.22, P = 0.005). Comparison of the mean AO across tertiles of lifestyle passivity scores showed onset 4.6 years (95% CI = 1.3-7.9) later in the least compared with the most passive tertile. CAG repeat length was also shown to predict lifestyle passivity (R(2) = 0.12, b = 1.08, P < 0.0005). Neither intellectual nor physical activity showed significant relationships to AO or CAG repeat length in this cohort. Our study leads to two conclusions: that a passive lifestyle may be a preclinical expression of HD, and that it actually contributes to the earlier onset of symptoms. Overcoming the tendency to be passive may substantially delay onset of HD. (c) 2010 Movement Disorder Society.
DOI: 10.1002/mds.23108
PubMed: 20629137
Affiliations:
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Horton</name></author><author><name sortKey="Tippett, Lynette" sort="Tippett, Lynette" uniqKey="Tippett L" first="Lynette" last="Tippett">Lynette Tippett</name></author><author><name sortKey="Hogg, Virginia" sort="Hogg, Virginia" uniqKey="Hogg V" first="Virginia" last="Hogg">Virginia Hogg</name></author><author><name sortKey="Collins, Veronica R" sort="Collins, Veronica R" uniqKey="Collins V" first="Veronica R" last="Collins">Veronica R. Collins</name></author><author><name sortKey="Churchyard, Andrew" sort="Churchyard, Andrew" uniqKey="Churchyard A" first="Andrew" last="Churchyard">Andrew Churchyard</name></author><author><name sortKey="Velakoulis, Dennis" sort="Velakoulis, Dennis" uniqKey="Velakoulis D" first="Dennis" last="Velakoulis">Dennis Velakoulis</name></author><author><name sortKey="Roxburgh, Richard" sort="Roxburgh, Richard" uniqKey="Roxburgh R" first="Richard" last="Roxburgh">Richard Roxburgh</name></author><author><name sortKey="Delatycki, Martin B" sort="Delatycki, Martin B" uniqKey="Delatycki M" first="Martin B" last="Delatycki">Martin B. 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Horton</name></author><author><name sortKey="Tippett, Lynette" sort="Tippett, Lynette" uniqKey="Tippett L" first="Lynette" last="Tippett">Lynette Tippett</name></author><author><name sortKey="Hogg, Virginia" sort="Hogg, Virginia" uniqKey="Hogg V" first="Virginia" last="Hogg">Virginia Hogg</name></author><author><name sortKey="Collins, Veronica R" sort="Collins, Veronica R" uniqKey="Collins V" first="Veronica R" last="Collins">Veronica R. Collins</name></author><author><name sortKey="Churchyard, Andrew" sort="Churchyard, Andrew" uniqKey="Churchyard A" first="Andrew" last="Churchyard">Andrew Churchyard</name></author><author><name sortKey="Velakoulis, Dennis" sort="Velakoulis, Dennis" uniqKey="Velakoulis D" first="Dennis" last="Velakoulis">Dennis Velakoulis</name></author><author><name sortKey="Roxburgh, Richard" sort="Roxburgh, Richard" uniqKey="Roxburgh R" first="Richard" last="Roxburgh">Richard Roxburgh</name></author><author><name sortKey="Delatycki, Martin B" sort="Delatycki, Martin B" uniqKey="Delatycki M" first="Martin B" last="Delatycki">Martin B. Delatycki</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="2010" type="published">2010</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Age of Onset</term><term>Aged</term><term>Cohort Studies</term><term>Female</term><term>Humans</term><term>Huntington Disease (epidemiology)</term><term>Huntington Disease (genetics)</term><term>Huntington Disease (psychology)</term><term>Life Style</term><term>Linear Models</term><term>Male</term><term>Middle Aged</term><term>Nerve Tissue Proteins (genetics)</term><term>New Zealand (epidemiology)</term><term>Nuclear Proteins (genetics)</term><term>Retrospective Studies</term><term>Statistics as Topic</term><term>Trinucleotide Repeat Expansion (genetics)</term><term>Young Adult</term></keywords><keywords scheme="MESH" type="chemical" qualifier="genetics" xml:lang="en"><term>Nerve Tissue Proteins</term><term>Nuclear Proteins</term></keywords><keywords scheme="MESH" type="geographic" qualifier="epidemiology" xml:lang="en"><term>New Zealand</term></keywords><keywords scheme="MESH" qualifier="epidemiology" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Huntington Disease</term><term>Trinucleotide Repeat Expansion</term></keywords><keywords scheme="MESH" qualifier="psychology" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Age of Onset</term><term>Aged</term><term>Cohort Studies</term><term>Female</term><term>Humans</term><term>Life Style</term><term>Linear Models</term><term>Male</term><term>Middle Aged</term><term>Retrospective Studies</term><term>Statistics as Topic</term><term>Young Adult</term></keywords><keywords scheme="Wicri" type="geographic" xml:lang="fr"><term>Nouvelle-Zélande</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">In transgenic mouse models of Huntington disease (HD) environmental enrichment significantly delays disease onset. A questionnaire-based survey of 154 adults with diagnosed HD (mean 4.2 years postdiagnosis) and a known IT15 CAG repeat length, explored whether premorbid lifestyle may relate to age-at-onset (AO). Participants were drawn from HD outpatient clinics in Australia and New Zealand. Premorbid physical, intellectual, and passive activity levels were used to generate scores in the categories of leisure, nonleisure (education, occupation and domestic duties) and total lifestyle. AO was associated with increased CAG repeat length as expected (r = -0.72, P < 0.001), but also with a lifestyle that included higher levels of passive activity (r = -0.38, P < 0.001). Multiple linear regression modeling showed lifestyle passivity to be a variable independent of CAG repeat length in predicting AO (R(2) = 0.54, b = -0.22, P = 0.005). Comparison of the mean AO across tertiles of lifestyle passivity scores showed onset 4.6 years (95% CI = 1.3-7.9) later in the least compared with the most passive tertile. CAG repeat length was also shown to predict lifestyle passivity (R(2) = 0.12, b = 1.08, P < 0.0005). Neither intellectual nor physical activity showed significant relationships to AO or CAG repeat length in this cohort. Our study leads to two conclusions: that a passive lifestyle may be a preclinical expression of HD, and that it actually contributes to the earlier onset of symptoms. Overcoming the tendency to be passive may substantially delay onset of HD. (c) 2010 Movement Disorder Society.</div></front></TEI><affiliations><list><country><li>Australie</li></country></list><tree><noCountry><name sortKey="Churchyard, Andrew" sort="Churchyard, Andrew" uniqKey="Churchyard A" first="Andrew" last="Churchyard">Andrew Churchyard</name><name sortKey="Collins, Veronica R" sort="Collins, Veronica R" uniqKey="Collins V" first="Veronica R" last="Collins">Veronica R. Collins</name><name sortKey="Delatycki, Martin B" sort="Delatycki, Martin B" uniqKey="Delatycki M" first="Martin B" last="Delatycki">Martin B. Delatycki</name><name sortKey="Hogg, Virginia" sort="Hogg, Virginia" uniqKey="Hogg V" first="Virginia" last="Hogg">Virginia Hogg</name><name sortKey="Horton, Zoe A" sort="Horton, Zoe A" uniqKey="Horton Z" first="Zoë A" last="Horton">Zoë A. Horton</name><name sortKey="Roxburgh, Richard" sort="Roxburgh, Richard" uniqKey="Roxburgh R" first="Richard" last="Roxburgh">Richard Roxburgh</name><name sortKey="Tippett, Lynette" sort="Tippett, Lynette" uniqKey="Tippett L" first="Lynette" last="Tippett">Lynette Tippett</name><name sortKey="Velakoulis, Dennis" sort="Velakoulis, Dennis" uniqKey="Velakoulis D" first="Dennis" last="Velakoulis">Dennis Velakoulis</name></noCountry><country name="Australie"><noRegion><name sortKey="Trembath, M Kaye" sort="Trembath, M Kaye" uniqKey="Trembath M" first="M Kaye" last="Trembath">M Kaye Trembath</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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