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<meta name="DC.Title" content="Topological Alterations of the Structural Brain Connectivity Network in Children with Juvenile Neuronal Ceroid Lipofuscinosis" />
<meta name="DC.Identifier" content="10.3174/ajnr.A6306" />
<meta name="DC.Date" content="2019-12-01" />
<meta name="DC.Publisher" content="American Journal of Neuroradiology" />
<meta name="DC.Rights" content="© 2019 by American Journal of Neuroradiology. Indicates open access to non-subscribers at www.ajnr.org" />
<meta name="DC.AccessRights" content="open-access" />
<meta name="DC.Description" content="BACKGROUND AND PURPOSE: We used diffusion MR imaging to investigate the structural brain connectivity networks in juvenile neuronal ceroid lipofuscinosis, a neurodegenerative lysosomal storage disease of childhood. Although changes in conventional MR imaging are typically not visually apparent in children aged &lt;10 years, we previously found significant microstructural abnormalities by using diffusion MR imaging. Therefore, we hypothesized that the structural connectivity networks would also be affected in the disease.

MATERIALS AND METHODS: We acquired diffusion MR imaging data from 14 children with juvenile neuronal ceroid lipofuscinosis (mean ± SD age, 9.6 ± 3.4 years; 10 boys) and 14 control subjects (mean ± SD age, 11.2 ± 2.3 years; 7 boys). A follow-up MR imaging was performed for 12 of the patients (mean ± SD age, 11.4 ± 3.2 years; 8 boys). We used graph theoretical analysis to investigate the global and local properties of the structural brain connectivity networks reconstructed with constrained spherical deconvolution–based whole-brain probabilistic tractography.

RESULTS: We found significantly increased characteristic path length ( P  = .003) and decreased degree ( P  = .003), which indicated decreased network integration and centrality in children with juvenile neuronal ceroid lipofuscinosis. The findings were similar for the follow-up MR imaging, and there were no significant differences between the two acquisitions of the patients. In addition, we found that the disease severity correlated negatively ( P  &lt; .007) with integration, segregation, centrality, and small-worldness of the networks. Moreover, we found significantly ( P  &lt; .0003) decreased local efficiency in the left supramarginal gyrus and temporal plane, and decreased strength in the right lingual gyrus.

CONCLUSIONS: We found significant global and local network alterations in juvenile neuronal ceroid lipofuscinosis that correlated with the disease severity and in areas related to the symptomatology.

CLN3
:   juvenile neuronal ceroid lipofuscinosis
CSD
:   constrained spherical deconvolution
LSD
:   lysosomal storage disease
UPDRS
:   Unified Parkinson’s Disease Rating Scale" />
<meta name="DC.Contributor" content="T. Roine" />
<meta name="DC.Contributor" content="U. Roine" />
<meta name="DC.Contributor" content="A. Tokola" />
<meta name="DC.Contributor" content="M.H. Balk" />
<meta name="DC.Contributor" content="M. Mannerkoski" />
<meta name="DC.Contributor" content="L. Åberg" />
<meta name="DC.Contributor" content="T. Lönnqvist" />
<meta name="DC.Contributor" content="T. Autti" />
<meta name="article:published_time" content="2019-12-01" />
<meta name="article:section" content="Pediatrics" />
<meta name="citation_title" content="Topological Alterations of the Structural Brain Connectivity Network in Children with Juvenile Neuronal Ceroid Lipofuscinosis" />
<meta name="citation_abstract" lang="en" content="&lt;h3&gt;BACKGROUND AND PURPOSE:&lt;/h3&gt;
&lt;p&gt;We used diffusion MR imaging to investigate the structural brain connectivity networks in juvenile neuronal ceroid lipofuscinosis, a neurodegenerative lysosomal storage disease of childhood. Although changes in conventional MR imaging are typically not visually apparent in children aged &amp;lt;10 years, we previously found significant microstructural abnormalities by using diffusion MR imaging. Therefore, we hypothesized that the structural connectivity networks would also be affected in the disease.&lt;/p&gt;&lt;h3&gt;MATERIALS AND METHODS:&lt;/h3&gt;
&lt;p&gt;We acquired diffusion MR imaging data from 14 children with juvenile neuronal ceroid lipofuscinosis (mean ± SD age, 9.6 ± 3.4 years; 10 boys) and 14 control subjects (mean ± SD age, 11.2 ± 2.3 years; 7 boys). A follow-up MR imaging was performed for 12 of the patients (mean ± SD age, 11.4 ± 3.2 years; 8 boys). We used graph theoretical analysis to investigate the global and local properties of the structural brain connectivity networks reconstructed with constrained spherical deconvolution–based whole-brain probabilistic tractography.&lt;/p&gt;&lt;h3&gt;RESULTS:&lt;/h3&gt;
&lt;p&gt;We found significantly increased characteristic path length (&lt;i&gt;P &lt;/i&gt;= .003) and decreased degree (&lt;i&gt;P &lt;/i&gt;= .003), which indicated decreased network integration and centrality in children with juvenile neuronal ceroid lipofuscinosis. The findings were similar for the follow-up MR imaging, and there were no significant differences between the two acquisitions of the patients. In addition, we found that the disease severity correlated negatively (&lt;i&gt;P &lt;/i&gt;&amp;lt; .007) with integration, segregation, centrality, and small-worldness of the networks. Moreover, we found significantly (&lt;i&gt;P &lt;/i&gt;&amp;lt; .0003) decreased local efficiency in the left supramarginal gyrus and temporal plane, and decreased strength in the right lingual gyrus.&lt;/p&gt;&lt;h3&gt;CONCLUSIONS:&lt;/h3&gt;
&lt;p&gt;We found significant global and local network alterations in juvenile neuronal ceroid lipofuscinosis that correlated with the disease severity and in areas related to the symptomatology.&lt;/p&gt;" />
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<meta name="citation_author_institution" content="eDepartment of Neuroscience and Biomedical Engineering (T.R.), Aalto University School of Science, Espoo, Finland" />
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<meta name="twitter:title" content="Topological Alterations of the Structural Brain Connectivity Network in Children with Juvenile Neuronal Ceroid Lipofuscinosis" />
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<meta name="twitter:description" content="BACKGROUND AND PURPOSE: We used diffusion MR imaging to investigate the structural brain connectivity networks in juvenile neuronal ceroid lipofuscinosis, a neurodegenerative lysosomal storage disease of childhood. Although changes in conventional MR imaging are typically not visually apparent in children aged &lt;10 years, we previously found significant microstructural abnormalities by using diffusion MR imaging. Therefore, we hypothesized that the structural connectivity networks would also be affected in the disease.

MATERIALS AND METHODS: We acquired diffusion MR imaging data from 14 children with juvenile neuronal ceroid lipofuscinosis (mean ± SD age, 9.6 ± 3.4 years; 10 boys) and 14 control subjects (mean ± SD age, 11.2 ± 2.3 years; 7 boys). A follow-up MR imaging was performed for 12 of the patients (mean ± SD age, 11.4 ± 3.2 years; 8 boys). We used graph theoretical analysis to investigate the global and local properties of the structural brain connectivity networks reconstructed with constrained spherical deconvolution–based whole-brain probabilistic tractography.

RESULTS: We found significantly increased characteristic path length ( P  = .003) and decreased degree ( P  = .003), which indicated decreased network integration and centrality in children with juvenile neuronal ceroid lipofuscinosis. The findings were similar for the follow-up MR imaging, and there were no significant differences between the two acquisitions of the patients. In addition, we found that the disease severity correlated negatively ( P  &lt; .007) with integration, segregation, centrality, and small-worldness of the networks. Moreover, we found significantly ( P  &lt; .0003) decreased local efficiency in the left supramarginal gyrus and temporal plane, and decreased strength in the right lingual gyrus.

CONCLUSIONS: We found significant global and local network alterations in juvenile neuronal ceroid lipofuscinosis that correlated with the disease severity and in areas related to the symptomatology.

CLN3
:   juvenile neuronal ceroid lipofuscinosis
CSD
:   constrained spherical deconvolution
LSD
:   lysosomal storage disease
UPDRS
:   Unified Parkinson’s Disease Rating Scale" />
<meta name="og-title" property="og:title" content="Topological Alterations of the Structural Brain Connectivity Network in Children with Juvenile Neuronal Ceroid Lipofuscinosis" />
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<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: We used diffusion MR imaging to investigate the structural brain connectivity networks in juvenile neuronal ceroid lipofuscinosis, a neurodegenerative lysosomal storage disease of childhood. Although changes in conventional MR imaging are typically not visually apparent in children aged &lt;10 years, we previously found significant microstructural abnormalities by using diffusion MR imaging. Therefore, we hypothesized that the structural connectivity networks would also be affected in the disease.

MATERIALS AND METHODS: We acquired diffusion MR imaging data from 14 children with juvenile neuronal ceroid lipofuscinosis (mean ± SD age, 9.6 ± 3.4 years; 10 boys) and 14 control subjects (mean ± SD age, 11.2 ± 2.3 years; 7 boys). A follow-up MR imaging was performed for 12 of the patients (mean ± SD age, 11.4 ± 3.2 years; 8 boys). We used graph theoretical analysis to investigate the global and local properties of the structural brain connectivity networks reconstructed with constrained spherical deconvolution–based whole-brain probabilistic tractography.

RESULTS: We found significantly increased characteristic path length ( P  = .003) and decreased degree ( P  = .003), which indicated decreased network integration and centrality in children with juvenile neuronal ceroid lipofuscinosis. The findings were similar for the follow-up MR imaging, and there were no significant differences between the two acquisitions of the patients. In addition, we found that the disease severity correlated negatively ( P  &lt; .007) with integration, segregation, centrality, and small-worldness of the networks. Moreover, we found significantly ( P  &lt; .0003) decreased local efficiency in the left supramarginal gyrus and temporal plane, and decreased strength in the right lingual gyrus.

CONCLUSIONS: We found significant global and local network alterations in juvenile neuronal ceroid lipofuscinosis that correlated with the disease severity and in areas related to the symptomatology.

CLN3
:   juvenile neuronal ceroid lipofuscinosis
CSD
:   constrained spherical deconvolution
LSD
:   lysosomal storage disease
UPDRS
:   Unified Parkinson’s Disease Rating Scale" />
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    	<div class="highwire-cite-authors"><span  class="highwire-citation-authors"><span class="highwire-citation-author first" data-delta="0">T. Roine</span>, <span class="highwire-citation-author" data-delta="1">U. Roine</span>, <span class="highwire-citation-author" data-delta="2">A. Tokola</span>, <span class="highwire-citation-author" data-delta="3">M.H. Balk</span>, <span class="highwire-citation-author" data-delta="4">M. Mannerkoski</span>, <span class="highwire-citation-author" data-delta="5">L. Åberg</span>, <span class="highwire-citation-author" data-delta="6">T. Lönnqvist</span> and <span class="highwire-citation-author" data-delta="7">T. Autti</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">December 2019,  </span><span  class="highwire-cite-metadata-volume highwire-cite-metadata">40 </span><span  class="highwire-cite-metadata-issue highwire-cite-metadata">(12) </span><span  class="highwire-cite-metadata-pages highwire-cite-metadata">2146-2153; </span><span  class="highwire-cite-metadata-doi highwire-cite-metadata">DOI: https://doi.org/10.3174/ajnr.A6306 </span></div>
  
  
  
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<div id="hw-article-author-popups-top-node-71905--6820871027" style="display: none;"><div class="author-tooltip-0"><div class="author-tooltip-name">T. Roine </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>b</span>Radiology, Child Psychiatry (M.M.)</div><div class='author-affiliation'><span class='nlm-sup'>d</span>Turku Brain and Mind Center (T.R.), University of Turku, Turku, Finland</div><div class='author-affiliation'><span class='nlm-sup'>e</span>Department of Neuroscience and Biomedical Engineering (T.R.), Aalto University School of Science, Espoo, Finland</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=T.%2BRoine%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=Roine%20T&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"><a href="/search/author1%3AT.%2BRoine%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li><li class="author-orcid-link last"><a href="https://orcid.org/0000-0002-3104-9734" target="_blank" class="" data-icon-position="" data-hide-link-title="0">ORCID record for T. Roine</a></li></ul></div><div class="author-tooltip-1"><div class="author-tooltip-name">U. Roine </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>b</span>Radiology, Child Psychiatry (M.M.)</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=U.%2BRoine%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=Roine%20U&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"><a href="/search/author1%3AU.%2BRoine%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li><li class="author-orcid-link last"><a href="https://orcid.org/0000-0002-6985-4261" target="_blank" class="" data-icon-position="" data-hide-link-title="0">ORCID record for U. Roine</a></li></ul></div><div class="author-tooltip-2"><div class="author-tooltip-name">A. Tokola </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>b</span>Radiology, Child Psychiatry (M.M.)</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=A.%2BTokola%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=Tokola%20A&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"><a href="/search/author1%3AA.%2BTokola%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li><li class="author-orcid-link last"><a href="https://orcid.org/0000-0002-5135-2191" target="_blank" class="" data-icon-position="" data-hide-link-title="0">ORCID record for A. Tokola</a></li></ul></div><div class="author-tooltip-3"><div class="author-tooltip-name">M.H. Balk </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>b</span>Radiology, Child Psychiatry (M.M.)</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=M.H.%2BBalk%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=Balk%20M&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"><a href="/search/author1%3AM.H.%2BBalk%2B" rel="nofollow" class="" data-icon-position="" data-hide-link-title="0">Search for this author on this site</a></li><li class="author-orcid-link last"><a href="https://orcid.org/0000-0002-6573-246X" target="_blank" class="" data-icon-position="" data-hide-link-title="0">ORCID record for M.H. Balk</a></li></ul></div><div class="author-tooltip-4"><div class="author-tooltip-name">M. 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    	<div class="highwire-cite-metadata"><span  class="highwire-cite-metadata-oa-ind highwire-cite-metadata"><i class="highwire-oa-indicator"></i> </span><span  class="highwire-cite-metadata-journal highwire-cite-metadata">American Journal of Neuroradiology </span><span  class="highwire-cite-metadata-date highwire-cite-metadata">Dec 2019, </span><span  class="highwire-cite-metadata-volume highwire-cite-metadata">40 </span><span  class="highwire-cite-metadata-issue highwire-cite-metadata">(12) </span><span  class="highwire-cite-metadata-pages highwire-cite-metadata">2146-2153; </span><span  class="highwire-cite-metadata-doi highwire-cite-metadata"><span class="label">DOI:</span> 10.3174/ajnr.A6306 </span></div>
  
  
  
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      <div class="highwire-cite-title">Topological Alterations of the Structural Brain Connectivity Network in Children with Juvenile Neuronal Ceroid Lipofuscinosis</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">T.</span> <span class="nlm-surname">Roine</span></span>, <span class="highwire-citation-author" data-delta="1"><span class="nlm-given-names">U.</span> <span class="nlm-surname">Roine</span></span>, <span class="highwire-citation-author" data-delta="2"><span class="nlm-given-names">A.</span> <span class="nlm-surname">Tokola</span></span>, <span class="highwire-citation-author" data-delta="3"><span class="nlm-given-names">M.H.</span> <span class="nlm-surname">Balk</span></span>, <span class="highwire-citation-author" data-delta="4"><span class="nlm-given-names">M.</span> <span class="nlm-surname">Mannerkoski</span></span>, <span class="highwire-citation-author" data-delta="5"><span class="nlm-given-names">L.</span> <span class="nlm-surname">Åberg</span></span>, <span class="highwire-citation-author" data-delta="6"><span class="nlm-given-names">T.</span> <span class="nlm-surname">Lönnqvist</span></span>, <span class="highwire-citation-author" data-delta="7"><span class="nlm-given-names">T.</span> <span class="nlm-surname">Autti</span></span></span></div>
  
    	<div class="highwire-cite-metadata"><span  class="highwire-cite-metadata-oa-ind highwire-cite-metadata"><i class="highwire-oa-indicator"></i> </span><span  class="highwire-cite-metadata-journal highwire-cite-metadata">American Journal of Neuroradiology </span><span  class="highwire-cite-metadata-date highwire-cite-metadata">Dec 2019, </span><span  class="highwire-cite-metadata-volume highwire-cite-metadata">40 </span><span  class="highwire-cite-metadata-issue highwire-cite-metadata">(12) </span><span  class="highwire-cite-metadata-pages highwire-cite-metadata">2146-2153; </span><span  class="highwire-cite-metadata-doi highwire-cite-metadata"><span class="label">DOI:</span> 10.3174/ajnr.A6306 </span></div>
  
  
  
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