Altered corticomotor-cerebellar integrity in young ataxia telangiectasia patients.
Identifieur interne : 000666 ( Main/Exploration ); précédent : 000665; suivant : 000667Altered corticomotor-cerebellar integrity in young ataxia telangiectasia patients.
Auteurs : Ishani Sahama [Australie] ; Kate Sinclair ; Simona Fiori ; Kerstin Pannek ; Martin Lavin ; Stephen RoseSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2014.
English descriptors
- KwdEn :
- Adolescent, Anisotropy, Ataxia Telangiectasia (pathology), Case-Control Studies, Cerebellum (pathology), Child, Diffusion Magnetic Resonance Imaging, Eye Movements (physiology), Female, Humans, Image Processing, Computer-Assisted, Magnetic Resonance Imaging, Male, Motor Cortex (pathology), Neural Pathways (pathology), Severity of Illness Index, White Matter (pathology), Young Adult.
- MESH :
- pathology : Ataxia Telangiectasia, Cerebellum, Motor Cortex, Neural Pathways, White Matter.
- physiology : Eye Movements.
- Adolescent, Anisotropy, Case-Control Studies, Child, Diffusion Magnetic Resonance Imaging, Female, Humans, Image Processing, Computer-Assisted, Magnetic Resonance Imaging, Male, Severity of Illness Index, Young Adult.
Abstract
Magnetic resonance imaging (MRI) research in identifying altered brain structure and function in ataxia-telangiectasia, an autosomal recessive neurodegenerative disorder, is limited. Diffusion-weighted MRI were obtained from 11 ataxia telangiectasia patients (age range, 7-22 years; mean, 12 years) and 11 typically developing age-matched participants (age range, 8-23 years; mean, 13 years). Gray matter volume alterations in patients were compared with those of healthy controls using voxel-based morphometry, whereas tract-based spatial statistics was employed to elucidate white matter microstructure differences between groups. White matter microstructure was probed using quantitative fractional anisotropy and mean diffusivity measures. Reduced gray matter volume in both cerebellar hemispheres and in the precentral-postcentral gyrus in the left cerebral hemisphere was observed in ataxia telangiectasia patients compared with controls (P < 0.05, corrected for multiple comparisons). A significant reduction in fractional anisotropy in the cerebellar hemispheres, anterior/posterior horns of the medulla, cerebral peduncles, and internal capsule white matter, particularly in the left posterior limb of the internal capsule and corona radiata in the left cerebral hemisphere, was observed in patients compared with controls (P < 0.05). Mean diffusivity differences were observed within the left cerebellar hemisphere and the white matter of the superior lobule of the right cerebellar hemisphere (P < 0.05). Cerebellum-localized gray matter changes are seen in young ataxia telangiectasia patients along with white matter tract degeneration projecting from the cerebellum into corticomotor regions. The lack of cortical involvement may reflect early-stage white matter motor pathway degeneration within young patients.
DOI: 10.1002/mds.25970
PubMed: 25042086
Affiliations:
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Le document en format XML
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last="Pannek">Kerstin Pannek</name></author><author><name sortKey="Lavin, Martin" sort="Lavin, Martin" uniqKey="Lavin M" first="Martin" last="Lavin">Martin Lavin</name></author><author><name sortKey="Rose, Stephen" sort="Rose, Stephen" uniqKey="Rose S" first="Stephen" last="Rose">Stephen Rose</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2014">2014</date><idno type="RBID">pubmed:25042086</idno><idno type="pmid">25042086</idno><idno type="doi">10.1002/mds.25970</idno><idno type="wicri:Area/PubMed/Corpus">000452</idno><idno type="wicri:Area/PubMed/Curation">000452</idno><idno type="wicri:Area/PubMed/Checkpoint">000683</idno><idno type="wicri:Area/Ncbi/Merge">004051</idno><idno type="wicri:Area/Ncbi/Curation">004051</idno><idno type="wicri:Area/Ncbi/Checkpoint">004051</idno><idno type="wicri:Area/Main/Merge">000666</idno><idno type="wicri:Area/Main/Curation">000666</idno><idno 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sort="Pannek, Kerstin" uniqKey="Pannek K" first="Kerstin" last="Pannek">Kerstin Pannek</name></author><author><name sortKey="Lavin, Martin" sort="Lavin, Martin" uniqKey="Lavin M" first="Martin" last="Lavin">Martin Lavin</name></author><author><name sortKey="Rose, Stephen" sort="Rose, Stephen" uniqKey="Rose S" first="Stephen" last="Rose">Stephen Rose</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>Adolescent</term><term>Anisotropy</term><term>Ataxia Telangiectasia (pathology)</term><term>Case-Control Studies</term><term>Cerebellum (pathology)</term><term>Child</term><term>Diffusion Magnetic Resonance Imaging</term><term>Eye Movements (physiology)</term><term>Female</term><term>Humans</term><term>Image Processing, Computer-Assisted</term><term>Magnetic Resonance Imaging</term><term>Male</term><term>Motor Cortex (pathology)</term><term>Neural Pathways (pathology)</term><term>Severity of Illness Index</term><term>White Matter (pathology)</term><term>Young Adult</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Ataxia Telangiectasia</term><term>Cerebellum</term><term>Motor Cortex</term><term>Neural Pathways</term><term>White Matter</term></keywords><keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Eye Movements</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Anisotropy</term><term>Case-Control Studies</term><term>Child</term><term>Diffusion Magnetic Resonance Imaging</term><term>Female</term><term>Humans</term><term>Image Processing, Computer-Assisted</term><term>Magnetic Resonance Imaging</term><term>Male</term><term>Severity of Illness Index</term><term>Young Adult</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Magnetic resonance imaging (MRI) research in identifying altered brain structure and function in ataxia-telangiectasia, an autosomal recessive neurodegenerative disorder, is limited. Diffusion-weighted MRI were obtained from 11 ataxia telangiectasia patients (age range, 7-22 years; mean, 12 years) and 11 typically developing age-matched participants (age range, 8-23 years; mean, 13 years). Gray matter volume alterations in patients were compared with those of healthy controls using voxel-based morphometry, whereas tract-based spatial statistics was employed to elucidate white matter microstructure differences between groups. White matter microstructure was probed using quantitative fractional anisotropy and mean diffusivity measures. Reduced gray matter volume in both cerebellar hemispheres and in the precentral-postcentral gyrus in the left cerebral hemisphere was observed in ataxia telangiectasia patients compared with controls (P < 0.05, corrected for multiple comparisons). A significant reduction in fractional anisotropy in the cerebellar hemispheres, anterior/posterior horns of the medulla, cerebral peduncles, and internal capsule white matter, particularly in the left posterior limb of the internal capsule and corona radiata in the left cerebral hemisphere, was observed in patients compared with controls (P < 0.05). Mean diffusivity differences were observed within the left cerebellar hemisphere and the white matter of the superior lobule of the right cerebellar hemisphere (P < 0.05). Cerebellum-localized gray matter changes are seen in young ataxia telangiectasia patients along with white matter tract degeneration projecting from the cerebellum into corticomotor regions. The lack of cortical involvement may reflect early-stage white matter motor pathway degeneration within young patients.</div></front></TEI><affiliations><list><country><li>Australie</li></country></list><tree><noCountry><name sortKey="Fiori, Simona" sort="Fiori, Simona" uniqKey="Fiori S" first="Simona" last="Fiori">Simona Fiori</name><name sortKey="Lavin, Martin" sort="Lavin, Martin" uniqKey="Lavin M" first="Martin" last="Lavin">Martin Lavin</name><name sortKey="Pannek, Kerstin" sort="Pannek, Kerstin" uniqKey="Pannek K" first="Kerstin" last="Pannek">Kerstin Pannek</name><name sortKey="Rose, Stephen" sort="Rose, Stephen" uniqKey="Rose S" first="Stephen" last="Rose">Stephen Rose</name><name sortKey="Sinclair, Kate" sort="Sinclair, Kate" uniqKey="Sinclair K" first="Kate" last="Sinclair">Kate Sinclair</name></noCountry><country name="Australie"><noRegion><name sortKey="Sahama, Ishani" sort="Sahama, Ishani" uniqKey="Sahama I" first="Ishani" last="Sahama">Ishani Sahama</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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