Specific pattern of early white‐matter changes in pure hereditary spastic paraplegia
Identifieur interne : 000254 ( Istex/Corpus ); précédent : 000253; suivant : 000255Specific pattern of early white‐matter changes in pure hereditary spastic paraplegia
Auteurs : Thomas Duning ; Tobias Warnecke ; Anja Schirmacher ; Hagen Schiffbauer ; Hubertus Lohmann ; Siawoosh Mohammadi ; Peter Young ; Michael DeppeSource :
- Movement Disorders [ 0885-3185 ] ; 2010-09-15.
English descriptors
Abstract
Hereditary spastic paraplegias (HSP) are genetically and clinically heterogeneous neurodegenerative disorders. Most MR studies on HSP include very heterogeneous samples of patients, and findings were inconsistent. Here, we examined six patients with pure HSP and SPG4 mutations by clinical evaluation, detailed neuropsychological testing, and neuroimaging analyses, including conventional MRI, diffusion tensor imaging (DTI), and brain volumetry. Differences of voxel‐wise statistics and ROI‐based analysis of DTI data between patients and 32 healthy volunteers were evaluated. Although conventional MRI and brain volumetry were normal, DTI revealed widespread disturbance of white matter (WM) integrity (P < 0.001), mainly affecting the corticospinal tract. With longer disease duration, frontal regions were also involved. The WM changes were also present in subclinical subjects harbouring the pathogenic mutation. These subtle WM abnormalities have functional relevance because they correlated with clinical symptoms. Thus, early alterations of nerve fibres, which can be detected by DTI, might serve as a biological marker in HSP, in particular with respect to future longitudinal studies. © 2010 Movement Disorder Society
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DOI: 10.1002/mds.23211
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ISTEX:B48F1303FC73C43E53D344DE088EE63733FD5B9BLe document en format XML
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Most MR studies on HSP include very heterogeneous samples of patients, and findings were inconsistent. Here, we examined six patients with pure HSP and SPG4 mutations by clinical evaluation, detailed neuropsychological testing, and neuroimaging analyses, including conventional MRI, diffusion tensor imaging (DTI), and brain volumetry. Differences of voxel‐wise statistics and ROI‐based analysis of DTI data between patients and 32 healthy volunteers were evaluated. Although conventional MRI and brain volumetry were normal, DTI revealed widespread disturbance of white matter (WM) integrity (P < 0.001), mainly affecting the corticospinal tract. With longer disease duration, frontal regions were also involved. The WM changes were also present in subclinical subjects harbouring the pathogenic mutation. These subtle WM abnormalities have functional relevance because they correlated with clinical symptoms. Thus, early alterations of nerve fibres, which can be detected by DTI, might serve as a biological marker in HSP, in particular with respect to future longitudinal studies. © 2010 Movement Disorder Society</div></front></TEI><istex><corpusName>wiley</corpusName><author><json:item><name>Thomas Duning MD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string><json:string>Department of Neurology, University Hospital of Muenster, Albert‐Schweitzer‐Str. 33, 48149 Muenster, Germany</json:string></affiliations></json:item><json:item><name>Tobias Warnecke MD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Anja Schirmacher PhD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Hagen Schiffbauer MD</name><affiliations><json:string>Department of Clinical Radiology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Hubertus Lohmann PhD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Siawoosh Mohammadi PhD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Peter Young MD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item><json:item><name>Michael Deppe PhD</name><affiliations><json:string>Department of Neurology, University Hospital of Münster, Münster, Germany</json:string></affiliations></json:item></author><subject><json:item><lang><json:string>eng</json:string></lang><value>hereditary spastic paraplegia</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>diffusion tensor imaging</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>white‐matter changes</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>SPG4</value></json:item></subject><articleId><json:string>MDS23211</json:string></articleId><language><json:string>eng</json:string></language><originalGenre><json:string>article</json:string></originalGenre><abstract>Hereditary spastic paraplegias (HSP) are genetically and clinically heterogeneous neurodegenerative disorders. Most MR studies on HSP include very heterogeneous samples of patients, and findings were inconsistent. Here, we examined six patients with pure HSP and SPG4 mutations by clinical evaluation, detailed neuropsychological testing, and neuroimaging analyses, including conventional MRI, diffusion tensor imaging (DTI), and brain volumetry. Differences of voxel‐wise statistics and ROI‐based analysis of DTI data between patients and 32 healthy volunteers were evaluated. Although conventional MRI and brain volumetry were normal, DTI revealed widespread disturbance of white matter (WM) integrity (P > 0.001), mainly affecting the corticospinal tract. With longer disease duration, frontal regions were also involved. The WM changes were also present in subclinical subjects harbouring the pathogenic mutation. These subtle WM abnormalities have functional relevance because they correlated with clinical symptoms. 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xml:id="kwd4">SPG4</keyword></keywordGroup><abstractGroup><abstract type="main" xml:lang="en"><title type="main">Abstract</title><p>Hereditary spastic paraplegias (HSP) are genetically and clinically heterogeneous neurodegenerative disorders. Most MR studies on HSP include very heterogeneous samples of patients, and findings were inconsistent. Here, we examined six patients with pure HSP and SPG4 mutations by clinical evaluation, detailed neuropsychological testing, and neuroimaging analyses, including conventional MRI, diffusion tensor imaging (DTI), and brain volumetry. Differences of voxel‐wise statistics and ROI‐based analysis of DTI data between patients and 32 healthy volunteers were evaluated. Although conventional MRI and brain volumetry were normal, DTI revealed widespread disturbance of white matter (WM) integrity (<i>P</i> < 0.001), mainly affecting the corticospinal tract. With longer disease duration, frontal regions were also involved. The WM changes were also present in subclinical subjects harbouring the pathogenic mutation. These subtle WM abnormalities have functional relevance because they correlated with clinical symptoms. Thus, early alterations of nerve fibres, which can be detected by DTI, might serve as a biological marker in HSP, in particular with respect to future longitudinal studies. © 2010 Movement Disorder Society</p></abstract></abstractGroup></contentMeta><noteGroup><note xml:id="fn1"><p>Potential conflict of interest: Nothing to report.</p></note><note xml:id="fn2"><p>The first two authors contributed equally to this work.</p></note></noteGroup></header></component></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Specific pattern of early white‐matter changes in pure hereditary spastic paraplegia</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Specific DTI Changes in Pure HSP</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Specific pattern of early white‐matter changes in pure hereditary spastic paraplegia</title></titleInfo><name type="personal"><namePart type="given">Thomas</namePart><namePart type="family">Duning</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><description>The first two authors contributed equally to this work.</description><affiliation>Department of Neurology, University Hospital of Muenster, Albert‐Schweitzer‐Str. 33, 48149 Muenster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Tobias</namePart><namePart type="family">Warnecke</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><description>The first two authors contributed equally to this work.</description><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Anja</namePart><namePart type="family">Schirmacher</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Hagen</namePart><namePart type="family">Schiffbauer</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Clinical Radiology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Hubertus</namePart><namePart type="family">Lohmann</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Siawoosh</namePart><namePart type="family">Mohammadi</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Peter</namePart><namePart type="family">Young</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Michael</namePart><namePart type="family">Deppe</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Department of Neurology, University Hospital of Münster, Münster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="article" displayLabel="article"></genre><originInfo><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><place><placeTerm type="text">Hoboken</placeTerm></place><dateIssued encoding="w3cdtf">2010-09-15</dateIssued><dateCaptured encoding="w3cdtf">2009-12-21</dateCaptured><dateValid encoding="w3cdtf">2010-03-31</dateValid><copyrightDate encoding="w3cdtf">2010</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><physicalDescription><internetMediaType>text/html</internetMediaType><extent unit="figures">2</extent><extent unit="tables">1</extent><extent unit="references">18</extent><extent unit="words">4110</extent></physicalDescription><abstract lang="en">Hereditary spastic paraplegias (HSP) are genetically and clinically heterogeneous neurodegenerative disorders. Most MR studies on HSP include very heterogeneous samples of patients, and findings were inconsistent. Here, we examined six patients with pure HSP and SPG4 mutations by clinical evaluation, detailed neuropsychological testing, and neuroimaging analyses, including conventional MRI, diffusion tensor imaging (DTI), and brain volumetry. Differences of voxel‐wise statistics and ROI‐based analysis of DTI data between patients and 32 healthy volunteers were evaluated. Although conventional MRI and brain volumetry were normal, DTI revealed widespread disturbance of white matter (WM) integrity (P < 0.001), mainly affecting the corticospinal tract. With longer disease duration, frontal regions were also involved. The WM changes were also present in subclinical subjects harbouring the pathogenic mutation. These subtle WM abnormalities have functional relevance because they correlated with clinical symptoms. Thus, early alterations of nerve fibres, which can be detected by DTI, might serve as a biological marker in HSP, in particular with respect to future longitudinal studies. © 2010 Movement Disorder Society</abstract><note type="content">*Potential conflict of interest: Nothing to report.</note><subject lang="en"><genre>keywords</genre><topic>hereditary spastic paraplegia</topic><topic>diffusion tensor imaging</topic><topic>white‐matter changes</topic><topic>SPG4</topic></subject><relatedItem type="host"><titleInfo><title>Movement Disorders</title></titleInfo><titleInfo type="abbreviated"><title>Mov. Disord.</title></titleInfo><genre type="journal">journal</genre><subject><genre>article-category</genre><topic>Brief Report</topic></subject><identifier type="ISSN">0885-3185</identifier><identifier type="eISSN">1531-8257</identifier><identifier type="DOI">10.1002/(ISSN)1531-8257</identifier><identifier type="PublisherID">MDS</identifier><part><date>2010</date><detail type="volume"><caption>vol.</caption><number>25</number></detail><detail type="issue"><caption>no.</caption><number>12</number></detail><extent unit="pages"><start>1986</start><end>1992</end><total>7</total></extent></part></relatedItem><identifier type="istex">B48F1303FC73C43E53D344DE088EE63733FD5B9B</identifier><identifier type="DOI">10.1002/mds.23211</identifier><identifier type="ArticleID">MDS23211</identifier><accessCondition type="use and reproduction" contentType="copyright">Published 2010 Wiley‐Liss, Inc.</accessCondition><recordInfo><recordContentSource>WILEY</recordContentSource><recordOrigin>Wiley Subscription Services, Inc., A Wiley Company</recordOrigin></recordInfo></mods></metadata><serie></serie></istex></record>Pour manipuler ce document sous Unix (Dilib)
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