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<meta name="DC.Title" content="Neuroimaging Correlates with Clinical Severity in Wilson Disease: A Multiparametric Quantitative Brain MRI" />
<meta name="DC.Identifier" content="10.3174/ajnr.A8479" />
<meta name="DC.Date" content="2024-11-01" />
<meta name="DC.Publisher" content="American Journal of Neuroradiology" />
<meta name="DC.Rights" content="© 2024 by American Journal of Neuroradiology" />
<meta name="DC.AccessRights" content="restricted" />
<meta name="DC.Description" content="BACKGROUND AND PURPOSE: Previous studies have reported metal accumulation and microstructure changes in deep gray nuclei (DGN) in Wilson disease (WD). However, there are limited studies that investigate whether there is metal accumulation and microstructure changes in DGN of patients with WD with normal-appearing routine MRI. This study aimed to evaluate multiparametric changes in DGN of WD and whether the findings correlate with clinical severity in patients with WD.

MATERIALS AND METHODS: The study enrolled 28 patients with WD (19 with neurologic symptoms) and 25 controls. Fractional anisotropy (FA), mean diffusivity (MD), and magnetic susceptibility in globus pallidus, pontine tegmentum, dentate nucleus, red nucleus, head of caudate nucleus, putamen, substantia nigra, and thalamus were extracted. Correlations between imaging data and the Unified Wilson’s Disease Rating Scale (UWDRS) neurologic subitems were explored.

RESULTS: FA, MD, and susceptibility values were higher in multiple DGN of patients with WD than controls ( P &lt; .05). Patients with WD without abnormal signals in DGN on routine MRI also had higher FA, MD, and susceptibility values than controls ( P &lt; .017). We found that UWDRS neurologic subscores correlated with FA and susceptibility values of DGN ( P &lt; .05). In addition, we also found that FA and susceptibility values in specific structures correlated with specific neurologic symptoms of WD (ie, tremor, parkinsonism, dysarthria, dystonia, and ataxia) ( P &lt; .05).

CONCLUSIONS: Patients with WD have increased FA, MD, and susceptibility values even before the lesion is morphologically apparent on routine MRI. The increased FA and susceptibility values correlate with clinical severity of WD.

CN
:   head of the caudate nucleus
DGN
:   deep gray nuclei
DN
:   dentate nucleus
FA
:   fractional anisotropy
GP
:   globus pallidus
GRE
:   gradient echo
ICC
:   intraclass correlation coefficient
MD
:   mean diffusivity
PT
:   pontine tegmentum
Put
:   putamen
QSM
:   quantitative susceptibility mapping
RN
:   red nucleus
SN
:   substantia nigra
TH
:   thalamus
UWDRS
:   Unified Wilson’s Disease Rating Scale
WD
:   Wilson disease" />
<meta name="DC.Contributor" content="Xiao-Zhong Jing" />
<meta name="DC.Contributor" content="Gai-Ying Li" />
<meta name="DC.Contributor" content="Yu-Peng Wu" />
<meta name="DC.Contributor" content="Xiang-Zhen Yuan" />
<meta name="DC.Contributor" content="Jia-Lin Chen" />
<meta name="DC.Contributor" content="Shu-Hong Wang" />
<meta name="DC.Contributor" content="Xiao-Ping Wang" />
<meta name="DC.Contributor" content="Jian-Qi Li" />
<meta name="article:published_time" content="2024-11-01" />
<meta name="article:section" content="Neurodegenerative Disorder Imaging" />
<meta name="citation_title" content="Neuroimaging Correlates with Clinical Severity in Wilson Disease: A Multiparametric Quantitative Brain MRI" />
<meta name="citation_abstract" lang="en" content="&lt;h3&gt;BACKGROUND AND PURPOSE:&lt;/h3&gt;
&lt;p&gt;Previous studies have reported metal accumulation and microstructure changes in deep gray nuclei (DGN) in Wilson disease (WD). However, there are limited studies that investigate whether there is metal accumulation and microstructure changes in DGN of patients with WD with normal-appearing routine MRI. This study aimed to evaluate multiparametric changes in DGN of WD and whether the findings correlate with clinical severity in patients with WD.&lt;/p&gt;&lt;h3&gt;MATERIALS AND METHODS:&lt;/h3&gt;
&lt;p&gt;The study enrolled 28 patients with WD (19 with neurologic symptoms) and 25 controls. Fractional anisotropy (FA), mean diffusivity (MD), and magnetic susceptibility in globus pallidus, pontine tegmentum, dentate nucleus, red nucleus, head of caudate nucleus, putamen, substantia nigra, and thalamus were extracted. Correlations between imaging data and the Unified Wilson’s Disease Rating Scale (UWDRS) neurologic subitems were explored.&lt;/p&gt;&lt;h3&gt;RESULTS:&lt;/h3&gt;
&lt;p&gt;FA, MD, and susceptibility values were higher in multiple DGN of patients with WD than controls (&lt;i&gt;P&lt;/i&gt; &amp;lt; .05). Patients with WD without abnormal signals in DGN on routine MRI also had higher FA, MD, and susceptibility values than controls (&lt;i&gt;P&lt;/i&gt; &amp;lt; .017). We found that UWDRS neurologic subscores correlated with FA and susceptibility values of DGN (&lt;i&gt;P&lt;/i&gt; &amp;lt; .05). In addition, we also found that FA and susceptibility values in specific structures correlated with specific neurologic symptoms of WD (ie, tremor, parkinsonism, dysarthria, dystonia, and ataxia) (&lt;i&gt;P&lt;/i&gt; &amp;lt; .05).&lt;/p&gt;&lt;h3&gt;CONCLUSIONS:&lt;/h3&gt;
&lt;p&gt;Patients with WD have increased FA, MD, and susceptibility values even before the lesion is morphologically apparent on routine MRI. The increased FA and susceptibility values correlate with clinical severity of WD.&lt;/p&gt;" />
<meta name="citation_journal_title" content="American Journal of Neuroradiology" />
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<meta name="citation_publication_date" content="2024/11/01" />
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<meta name="twitter:title" content="Neuroimaging Correlates with Clinical Severity in Wilson Disease: A Multiparametric Quantitative Brain MRI" />
<meta name="twitter:card" content="summary_large_image" />
<meta name="twitter:image" content="http://ajnr-stage2.stage.highwire.org/sites/default/files/highwire/ajnr/45/11.cover-source.jpg" />
<meta name="twitter:description" content="BACKGROUND AND PURPOSE: Previous studies have reported metal accumulation and microstructure changes in deep gray nuclei (DGN) in Wilson disease (WD). However, there are limited studies that investigate whether there is metal accumulation and microstructure changes in DGN of patients with WD with normal-appearing routine MRI. This study aimed to evaluate multiparametric changes in DGN of WD and whether the findings correlate with clinical severity in patients with WD.

MATERIALS AND METHODS: The study enrolled 28 patients with WD (19 with neurologic symptoms) and 25 controls. Fractional anisotropy (FA), mean diffusivity (MD), and magnetic susceptibility in globus pallidus, pontine tegmentum, dentate nucleus, red nucleus, head of caudate nucleus, putamen, substantia nigra, and thalamus were extracted. Correlations between imaging data and the Unified Wilson’s Disease Rating Scale (UWDRS) neurologic subitems were explored.

RESULTS: FA, MD, and susceptibility values were higher in multiple DGN of patients with WD than controls ( P &lt; .05). Patients with WD without abnormal signals in DGN on routine MRI also had higher FA, MD, and susceptibility values than controls ( P &lt; .017). We found that UWDRS neurologic subscores correlated with FA and susceptibility values of DGN ( P &lt; .05). In addition, we also found that FA and susceptibility values in specific structures correlated with specific neurologic symptoms of WD (ie, tremor, parkinsonism, dysarthria, dystonia, and ataxia) ( P &lt; .05).

CONCLUSIONS: Patients with WD have increased FA, MD, and susceptibility values even before the lesion is morphologically apparent on routine MRI. The increased FA and susceptibility values correlate with clinical severity of WD.

CN
:   head of the caudate nucleus
DGN
:   deep gray nuclei
DN
:   dentate nucleus
FA
:   fractional anisotropy
GP
:   globus pallidus
GRE
:   gradient echo
ICC
:   intraclass correlation coefficient
MD
:   mean diffusivity
PT
:   pontine tegmentum
Put
:   putamen
QSM
:   quantitative susceptibility mapping
RN
:   red nucleus
SN
:   substantia nigra
TH
:   thalamus
UWDRS
:   Unified Wilson’s Disease Rating Scale
WD
:   Wilson disease" />
<meta name="og-title" property="og:title" content="Neuroimaging Correlates with Clinical Severity in Wilson Disease: A Multiparametric Quantitative Brain MRI" />
<meta name="og-url" property="og:url" content="http://ajnr-stage2.stage.highwire.org/content/45/11/1745" />
<meta name="og-site-name" property="og:site_name" content="American Journal of Neuroradiology" />
<meta name="og-description" property="og:description" content="BACKGROUND AND PURPOSE: Previous studies have reported metal accumulation and microstructure changes in deep gray nuclei (DGN) in Wilson disease (WD). However, there are limited studies that investigate whether there is metal accumulation and microstructure changes in DGN of patients with WD with normal-appearing routine MRI. This study aimed to evaluate multiparametric changes in DGN of WD and whether the findings correlate with clinical severity in patients with WD.

MATERIALS AND METHODS: The study enrolled 28 patients with WD (19 with neurologic symptoms) and 25 controls. Fractional anisotropy (FA), mean diffusivity (MD), and magnetic susceptibility in globus pallidus, pontine tegmentum, dentate nucleus, red nucleus, head of caudate nucleus, putamen, substantia nigra, and thalamus were extracted. Correlations between imaging data and the Unified Wilson’s Disease Rating Scale (UWDRS) neurologic subitems were explored.

RESULTS: FA, MD, and susceptibility values were higher in multiple DGN of patients with WD than controls ( P &lt; .05). Patients with WD without abnormal signals in DGN on routine MRI also had higher FA, MD, and susceptibility values than controls ( P &lt; .017). We found that UWDRS neurologic subscores correlated with FA and susceptibility values of DGN ( P &lt; .05). In addition, we also found that FA and susceptibility values in specific structures correlated with specific neurologic symptoms of WD (ie, tremor, parkinsonism, dysarthria, dystonia, and ataxia) ( P &lt; .05).

CONCLUSIONS: Patients with WD have increased FA, MD, and susceptibility values even before the lesion is morphologically apparent on routine MRI. The increased FA and susceptibility values correlate with clinical severity of WD.

CN
:   head of the caudate nucleus
DGN
:   deep gray nuclei
DN
:   dentate nucleus
FA
:   fractional anisotropy
GP
:   globus pallidus
GRE
:   gradient echo
ICC
:   intraclass correlation coefficient
MD
:   mean diffusivity
PT
:   pontine tegmentum
Put
:   putamen
QSM
:   quantitative susceptibility mapping
RN
:   red nucleus
SN
:   substantia nigra
TH
:   thalamus
UWDRS
:   Unified Wilson’s Disease Rating Scale
WD
:   Wilson disease" />
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      <div class="highwire-cite-overline"><span  class="highwire-cite-metadata-article-type highwire-cite-metadata">Research Article</span><span class="separator-pipe"></span><span  class="highwire-cite-metadata-art-cat highwire-cite-metadata"><span class="wrapper">Neurodegenerative Disorder Imaging</span></span></div>
  
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      <h1 class="highwire-cite-title" id="page-title">Neuroimaging Correlates with Clinical Severity in Wilson Disease: A Multiparametric Quantitative Brain MRI</h1>  
    	<div class="highwire-cite-authors"><span  class="highwire-citation-authors"><span class="highwire-citation-author first" data-delta="0">Xiao-Zhong Jing</span>, <span class="highwire-citation-author" data-delta="1">Gai-Ying Li</span>, <span class="highwire-citation-author" data-delta="2">Yu-Peng Wu</span>, <span class="highwire-citation-author" data-delta="3">Xiang-Zhen Yuan</span>, <span class="highwire-citation-author" data-delta="4">Jia-Lin Chen</span>, <span class="highwire-citation-author" data-delta="5">Shu-Hong Wang</span>, <span class="highwire-citation-author" data-delta="6">Xiao-Ping Wang</span> and <span class="highwire-citation-author" data-delta="7">Jian-Qi Li</span></span></div>
  
    	<div class="highwire-cite-metadata"><span  class="highwire-cite-metadata-journal highwire-cite-metadata">American Journal of Neuroradiology </span><span  class="highwire-cite-metadata-date highwire-cite-metadata">November 2024,  </span><span  class="highwire-cite-metadata-volume highwire-cite-metadata">45 </span><span  class="highwire-cite-metadata-issue highwire-cite-metadata">(11) </span><span  class="highwire-cite-metadata-pages highwire-cite-metadata">1745-1754; </span><span  class="highwire-cite-metadata-doi highwire-cite-metadata">DOI: https://doi.org/10.3174/ajnr.A8479 </span></div>
  
  
  
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<div id="hw-article-author-popups-top-node-111464--81028728750" style="display: none;"><div class="author-tooltip-0"><div class="author-tooltip-name">Xiao-Zhong Jing </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>1</span>From the Department of Neurology (X.-Z.J.), The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei, China</div><div class='author-affiliation'><span class='nlm-sup'>2</span>Department of Neurology (X.-Z.J.), Tongren Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Xiao-Zhong%2BJing%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google 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China Normal University, Shanghai, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Yu-Peng%2BWu%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Wu%20Y&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li class="author-site-search-link last"><a href="/search/author1%3AYu-Peng%2BWu%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li></ul></div><div class="author-tooltip-3"><div class="author-tooltip-name">Xiang-Zhen Yuan </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>4</span>Department of Neurology (X.-Z.Y.), Weifang People's Hospital, Weifang, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Xiang-Zhen%2BYuan%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Yuan%20X&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li class="author-site-search-link last"><a href="/search/author1%3AXiang-Zhen%2BYuan%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li></ul></div><div class="author-tooltip-4"><div class="author-tooltip-name">Jia-Lin Chen </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>3</span>Shanghai Key Laboratory of Magnetic Resonance (G.-Y.L., Y-P.W., J-L.C., J-Q.L.), School of Physics and Electronic Science, East China Normal University, Shanghai, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Jia-Lin%2BChen%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Chen%20J&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li class="author-site-search-link last"><a href="/search/author1%3AJia-Lin%2BChen%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li></ul></div><div class="author-tooltip-5"><div class="author-tooltip-name">Shu-Hong Wang </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>5</span>Department of Neurology (S.-H.W.), Shanghai East Hospital, School of Medicine, Tongji University, Shanghai, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Shu-Hong%2BWang%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Wang%20S&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li class="author-site-search-link last"><a href="/search/author1%3AShu-Hong%2BWang%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li></ul></div><div class="author-tooltip-6"><div class="author-tooltip-name">Xiao-Ping Wang </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>6</span>Department of Neurology (X.-P.W.), Jiading Branch of Shanghai General Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China</div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Xiao-Ping%2BWang%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Wang%20X&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" 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      <div class="highwire-cite-title">Neuroimaging in Wilson Disease Severity</div>  
    	<div class="highwire-cite-authors"><span  class="highwire-citation-authors"><span class="highwire-citation-author first" data-delta="0"><span class="nlm-given-names">Xiao-Zhong</span> <span class="nlm-surname">Jing</span></span>, <span class="highwire-citation-author" data-delta="1"><span class="nlm-given-names">Gai-Ying</span> <span class="nlm-surname">Li</span></span>, <span class="highwire-citation-author" data-delta="2"><span class="nlm-given-names">Yu-Peng</span> <span class="nlm-surname">Wu</span></span>, <span class="highwire-citation-author" data-delta="3"><span class="nlm-given-names">Xiang-Zhen</span> <span class="nlm-surname">Yuan</span></span>, <span class="highwire-citation-author" data-delta="4"><span class="nlm-given-names">Jia-Lin</span> <span class="nlm-surname">Chen</span></span>, <span class="highwire-citation-author" data-delta="5"><span class="nlm-given-names">Shu-Hong</span> <span class="nlm-surname">Wang</span></span>, <span class="highwire-citation-author" data-delta="6"><span class="nlm-given-names">Xiao-Ping</span> <span class="nlm-surname">Wang</span></span>, <span class="highwire-citation-author" data-delta="7"><span class="nlm-given-names">Jian-Qi</span> <span class="nlm-surname">Li</span></span></span></div>
  
    	<div class="highwire-cite-metadata"><span  class="highwire-cite-metadata-journal highwire-cite-metadata">American Journal of Neuroradiology </span><span  class="highwire-cite-metadata-date highwire-cite-metadata">Nov 2024, </span><span  class="highwire-cite-metadata-volume highwire-cite-metadata">45 </span><span  class="highwire-cite-metadata-issue highwire-cite-metadata">(11) </span><span  class="highwire-cite-metadata-pages highwire-cite-metadata">1745-1754; </span><span  class="highwire-cite-metadata-doi highwire-cite-metadata"><span class="label">DOI:</span> 10.3174/ajnr.A8479 </span></div>
  
  
  
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