Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease
Identifieur interne : 001936 ( Main/Exploration ); précédent : 001935; suivant : 001937Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease
Auteurs : Ralf Reilmann [Allemagne] ; Stefan Bohlen [Allemagne] ; Thomas Klopstock [Allemagne] ; Andreas Bender [Allemagne] ; Adolf Weindl [Allemagne] ; Philipp Saemann [Allemagne] ; Dorothee P. Auer [Allemagne] ; E. Bernd Ringelstein [Allemagne] ; Herwig W. Lange [Allemagne]Source :
- Movement Disorders [ 0885-3185 ] ; 2010-10-15.
Descripteurs français
- Pascal (Inist)
English descriptors
- KwdEn :
- Adult, Basal ganglion, Chorea, Disability Evaluation, Disease Progression, Female, Force, Genotype, Humans, Huntington Disease (pathology), Huntington Disease (physiopathology), Huntington disease, Huntington's disease, Male, Middle Aged, Motor control, Nervous system diseases, Phenotype, Predictive Value of Tests, Statistics, Nonparametric, Tongue, Tongue (physiopathology), Young Adult, basal ganglia, chorea, motor control.
- MESH :
- pathology : Huntington Disease.
- physiopathology : Huntington Disease, Tongue.
- Adult, Disability Evaluation, Disease Progression, Female, Genotype, Humans, Male, Middle Aged, Phenotype, Predictive Value of Tests, Statistics, Nonparametric, Young Adult.
Abstract
Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society
Url:
DOI: 10.1002/mds.23243
Affiliations:
Links toward previous steps (curation, corpus...)
- to stream Istex, to step Corpus: 000D80
- to stream Istex, to step Curation: 000D80
- to stream Istex, to step Checkpoint: 000615
- to stream PubMed, to step Corpus: 001707
- to stream PubMed, to step Curation: 001707
- to stream PubMed, to step Checkpoint: 001543
- to stream Ncbi, to step Merge: 002D10
- to stream Ncbi, to step Curation: 002D10
- to stream Ncbi, to step Checkpoint: 002D10
- to stream Main, to step Merge: 001D72
- to stream PascalFrancis, to step Corpus: 000891
- to stream PascalFrancis, to step Curation: 002428
- to stream PascalFrancis, to step Checkpoint: 000775
- to stream Main, to step Merge: 002362
- to stream Main, to step Curation: 001936
Le document en format XML
<record><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title><author><name sortKey="Reilmann, Ralf" sort="Reilmann, Ralf" uniqKey="Reilmann R" first="Ralf" last="Reilmann">Ralf Reilmann</name></author><author><name sortKey="Bohlen, Stefan" sort="Bohlen, Stefan" uniqKey="Bohlen S" first="Stefan" last="Bohlen">Stefan Bohlen</name></author><author><name sortKey="Klopstock, Thomas" sort="Klopstock, Thomas" uniqKey="Klopstock T" first="Thomas" last="Klopstock">Thomas Klopstock</name></author><author><name sortKey="Bender, Andreas" sort="Bender, Andreas" uniqKey="Bender A" first="Andreas" last="Bender">Andreas Bender</name></author><author><name sortKey="Weindl, Adolf" sort="Weindl, Adolf" uniqKey="Weindl A" first="Adolf" last="Weindl">Adolf Weindl</name></author><author><name sortKey="Saemann, Philipp" sort="Saemann, Philipp" uniqKey="Saemann P" first="Philipp" last="Saemann">Philipp Saemann</name></author><author><name sortKey="Auer, Dorothee P" sort="Auer, Dorothee P" uniqKey="Auer D" first="Dorothee P." last="Auer">Dorothee P. Auer</name></author><author><name sortKey="Ringelstein, E Bernd" sort="Ringelstein, E Bernd" uniqKey="Ringelstein E" first="E. Bernd" last="Ringelstein">E. Bernd Ringelstein</name></author><author><name sortKey="Lange, Herwig W" sort="Lange, Herwig W" uniqKey="Lange H" first="Herwig W." last="Lange">Herwig W. Lange</name></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:4340529D2182874F0A2B8EC8F45A449B0BDE6D06</idno><date when="2010" year="2010">2010</date><idno type="doi">10.1002/mds.23243</idno><idno type="url">https://api.istex.fr/document/4340529D2182874F0A2B8EC8F45A449B0BDE6D06/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">000D80</idno><idno type="wicri:Area/Istex/Curation">000D80</idno><idno type="wicri:Area/Istex/Checkpoint">000615</idno><idno type="wicri:doubleKey">0885-3185:2010:Reilmann R:tongue:force:analysis</idno><idno type="wicri:source">PubMed</idno><idno type="RBID">pubmed:20645403</idno><idno type="wicri:Area/PubMed/Corpus">001707</idno><idno type="wicri:Area/PubMed/Curation">001707</idno><idno type="wicri:Area/PubMed/Checkpoint">001543</idno><idno type="wicri:Area/Ncbi/Merge">002D10</idno><idno type="wicri:Area/Ncbi/Curation">002D10</idno><idno type="wicri:Area/Ncbi/Checkpoint">002D10</idno><idno type="wicri:Area/Main/Merge">001D72</idno><idno type="wicri:source">INIST</idno><idno type="RBID">Pascal:10-0474327</idno><idno type="wicri:Area/PascalFrancis/Corpus">000891</idno><idno type="wicri:Area/PascalFrancis/Curation">002428</idno><idno type="wicri:Area/PascalFrancis/Checkpoint">000775</idno><idno type="wicri:doubleKey">0885-3185:2010:Reilmann R:tongue:force:analysis</idno><idno type="wicri:Area/Main/Merge">002362</idno><idno type="wicri:Area/Main/Curation">001936</idno><idno type="wicri:Area/Main/Exploration">001936</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title><author><name sortKey="Reilmann, Ralf" sort="Reilmann, Ralf" uniqKey="Reilmann R" first="Ralf" last="Reilmann">Ralf Reilmann</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster</wicri:regionArea><wicri:noRegion>Muenster</wicri:noRegion><wicri:noRegion>Muenster</wicri:noRegion></affiliation></author><author><name sortKey="Bohlen, Stefan" sort="Bohlen, Stefan" uniqKey="Bohlen S" first="Stefan" last="Bohlen">Stefan Bohlen</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster</wicri:regionArea><wicri:noRegion>Muenster</wicri:noRegion><wicri:noRegion>Muenster</wicri:noRegion></affiliation></author><author><name sortKey="Klopstock, Thomas" sort="Klopstock, Thomas" uniqKey="Klopstock T" first="Thomas" last="Klopstock">Thomas Klopstock</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich</wicri:regionArea><placeName><region type="land" nuts="1">Bavière</region><region type="district" nuts="2">District de Haute-Bavière</region><settlement type="city">Munich</settlement></placeName></affiliation></author><author><name sortKey="Bender, Andreas" sort="Bender, Andreas" uniqKey="Bender A" first="Andreas" last="Bender">Andreas Bender</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich</wicri:regionArea><placeName><region type="land" nuts="1">Bavière</region><region type="district" nuts="2">District de Haute-Bavière</region><settlement type="city">Munich</settlement></placeName></affiliation></author><author><name sortKey="Weindl, Adolf" sort="Weindl, Adolf" uniqKey="Weindl A" first="Adolf" last="Weindl">Adolf Weindl</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University Munich, Munich</wicri:regionArea><placeName><region type="land" nuts="1">Bavière</region><region type="district" nuts="2">District de Haute-Bavière</region><settlement type="city">Munich</settlement></placeName></affiliation></author><author><name sortKey="Saemann, Philipp" sort="Saemann, Philipp" uniqKey="Saemann P" first="Philipp" last="Saemann">Philipp Saemann</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Radiology, Max Planck Institute of Psychiatry, Munich</wicri:regionArea><placeName><region type="land" nuts="1">Bavière</region><region type="district" nuts="2">District de Haute-Bavière</region><settlement type="city">Munich</settlement></placeName></affiliation></author><author><name sortKey="Auer, Dorothee P" sort="Auer, Dorothee P" uniqKey="Auer D" first="Dorothee P." last="Auer">Dorothee P. Auer</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Radiology, Max Planck Institute of Psychiatry, Munich</wicri:regionArea><placeName><region type="land" nuts="1">Bavière</region><region type="district" nuts="2">District de Haute-Bavière</region><settlement type="city">Munich</settlement></placeName></affiliation></author><author><name sortKey="Ringelstein, E Bernd" sort="Ringelstein, E Bernd" uniqKey="Ringelstein E" first="E. Bernd" last="Ringelstein">E. Bernd Ringelstein</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster</wicri:regionArea><wicri:noRegion>Muenster</wicri:noRegion><wicri:noRegion>Muenster</wicri:noRegion></affiliation></author><author><name sortKey="Lange, Herwig W" sort="Lange, Herwig W" uniqKey="Lange H" first="Herwig W." last="Lange">Herwig W. Lange</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster</wicri:regionArea><wicri:noRegion>Muenster</wicri:noRegion><wicri:noRegion>Muenster</wicri:noRegion></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. Disord.</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2010-10-15">2010-10-15</date><biblScope unit="vol">25</biblScope><biblScope unit="issue">13</biblScope><biblScope unit="page" from="2195">2195</biblScope><biblScope unit="page" to="2202">2202</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">4340529D2182874F0A2B8EC8F45A449B0BDE6D06</idno><idno type="DOI">10.1002/mds.23243</idno><idno type="ArticleID">MDS23243</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Basal ganglion</term><term>Chorea</term><term>Disability Evaluation</term><term>Disease Progression</term><term>Female</term><term>Force</term><term>Genotype</term><term>Humans</term><term>Huntington Disease (pathology)</term><term>Huntington Disease (physiopathology)</term><term>Huntington disease</term><term>Huntington's disease</term><term>Male</term><term>Middle Aged</term><term>Motor control</term><term>Nervous system diseases</term><term>Phenotype</term><term>Predictive Value of Tests</term><term>Statistics, Nonparametric</term><term>Tongue</term><term>Tongue (physiopathology)</term><term>Young Adult</term><term>basal ganglia</term><term>chorea</term><term>motor control</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Huntington Disease</term><term>Tongue</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Disability Evaluation</term><term>Disease Progression</term><term>Female</term><term>Genotype</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Phenotype</term><term>Predictive Value of Tests</term><term>Statistics, Nonparametric</term><term>Young Adult</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Chorée de Huntington</term><term>Contrôle moteur</term><term>Force</term><term>Langue</term><term>Noyau gris central</term><term>Pathologie du système nerveux</term><term>Phénotype</term><term>Syndrome choréique</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society</div></front></TEI><affiliations><list><country><li>Allemagne</li></country><region><li>Bavière</li><li>District de Haute-Bavière</li></region><settlement><li>Munich</li></settlement></list><tree><country name="Allemagne"><noRegion><name sortKey="Reilmann, Ralf" sort="Reilmann, Ralf" uniqKey="Reilmann R" first="Ralf" last="Reilmann">Ralf Reilmann</name></noRegion><name sortKey="Auer, Dorothee P" sort="Auer, Dorothee P" uniqKey="Auer D" first="Dorothee P." last="Auer">Dorothee P. Auer</name><name sortKey="Bender, Andreas" sort="Bender, Andreas" uniqKey="Bender A" first="Andreas" last="Bender">Andreas Bender</name><name sortKey="Bohlen, Stefan" sort="Bohlen, Stefan" uniqKey="Bohlen S" first="Stefan" last="Bohlen">Stefan Bohlen</name><name sortKey="Klopstock, Thomas" sort="Klopstock, Thomas" uniqKey="Klopstock T" first="Thomas" last="Klopstock">Thomas Klopstock</name><name sortKey="Lange, Herwig W" sort="Lange, Herwig W" uniqKey="Lange H" first="Herwig W." last="Lange">Herwig W. Lange</name><name sortKey="Ringelstein, E Bernd" sort="Ringelstein, E Bernd" uniqKey="Ringelstein E" first="E. Bernd" last="Ringelstein">E. Bernd Ringelstein</name><name sortKey="Saemann, Philipp" sort="Saemann, Philipp" uniqKey="Saemann P" first="Philipp" last="Saemann">Philipp Saemann</name><name sortKey="Weindl, Adolf" sort="Weindl, Adolf" uniqKey="Weindl A" first="Adolf" last="Weindl">Adolf Weindl</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
EXPLOR_STEP=$WICRI_ROOT/Wicri/Santé/explor/MovDisordV3/Data/Main/Exploration
HfdSelect -h $EXPLOR_STEP/biblio.hfd -nk 001936 | SxmlIndent | more
Ou
HfdSelect -h $EXPLOR_AREA/Data/Main/Exploration/biblio.hfd -nk 001936 | SxmlIndent | more
Pour mettre un lien sur cette page dans le réseau Wicri
{{Explor lien
|wiki= Wicri/Santé
|area= MovDisordV3
|flux= Main
|étape= Exploration
|type= RBID
|clé= ISTEX:4340529D2182874F0A2B8EC8F45A449B0BDE6D06
|texte= Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease
}}
| This area was generated with Dilib version V0.6.23. |  |