Disrupted Functional and Structural Connectivity in Angelman Syndrome
This work investigated alterations in functional connectivity (FC) and associated structures in patients with Angelman syndrome (AS) by using integrated quantitative imaging analysis and connectivity measures. We obtained 3T brain MR imaging, including resting-state functional MR imaging, diffusion...
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| Veröffentlicht in: | American journal of neuroradiology : AJNR 2020-05, Vol.41 (5), p.889-897 |
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| creator | Yoon, H M Jo, Y Shim, W H Lee, J S Ko, T S Koo, J H Yum, M S |
| description | This work investigated alterations in functional connectivity (FC) and associated structures in patients with Angelman syndrome (AS) by using integrated quantitative imaging analysis and connectivity measures.
We obtained 3T brain MR imaging, including resting-state functional MR imaging, diffusion tensor imaging, and 3D T1-weighted imaging from children with AS (
= 14) and age- and sex-matched controls (
= 28). The brains of patients with AS were analyzed by measuring FC, white matter microstructural analysis, cortical thickness, and brain volumes; these were compared with brains of controls.
Interregional FC analysis revealed significantly reduced intra- and interhemispheric FC, especially in the basal ganglia and thalamus, in patients with AS. Significant reductions in fractional anisotropy were found in the corpus callosum, cingulum, posterior limb of the internal capsules, and arcuate fasciculus in patients with AS. Quantitative structural analysis also showed gray matter volume loss of the basal ganglia and diffuse WM volume reduction in AS compared with the control group.
This integrated quantitative MR imaging analysis demonstrated poor functional and structural connectivity, as well as brain volume reduction, in children with AS, which may explain the motor and language dysfunction observed in this well-characterized neurobehavioral phenotype. |
| doi_str_mv | 10.3174/ajnr.A6531 |
| format | Article |
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We obtained 3T brain MR imaging, including resting-state functional MR imaging, diffusion tensor imaging, and 3D T1-weighted imaging from children with AS (
= 14) and age- and sex-matched controls (
= 28). The brains of patients with AS were analyzed by measuring FC, white matter microstructural analysis, cortical thickness, and brain volumes; these were compared with brains of controls.
Interregional FC analysis revealed significantly reduced intra- and interhemispheric FC, especially in the basal ganglia and thalamus, in patients with AS. Significant reductions in fractional anisotropy were found in the corpus callosum, cingulum, posterior limb of the internal capsules, and arcuate fasciculus in patients with AS. Quantitative structural analysis also showed gray matter volume loss of the basal ganglia and diffuse WM volume reduction in AS compared with the control group.
This integrated quantitative MR imaging analysis demonstrated poor functional and structural connectivity, as well as brain volume reduction, in children with AS, which may explain the motor and language dysfunction observed in this well-characterized neurobehavioral phenotype.</description><identifier>ISSN: 0195-6108</identifier><identifier>EISSN: 1936-959X</identifier><identifier>DOI: 10.3174/ajnr.A6531</identifier><identifier>PMID: 32381544</identifier><language>eng</language><publisher>United States: American Society of Neuroradiology</publisher><subject>Adolescent ; Angelman Syndrome - diagnostic imaging ; Angelman Syndrome - pathology ; Angelman Syndrome - physiopathology ; Brain - diagnostic imaging ; Brain - pathology ; Brain - physiopathology ; Child ; Child, Preschool ; Diffusion Tensor Imaging - methods ; Female ; Functional ; Humans ; Image Interpretation, Computer-Assisted - methods ; Male ; Nerve Net - diagnostic imaging ; Nerve Net - pathology ; Nerve Net - physiopathology ; Neuroimaging - methods ; Pediatrics</subject><ispartof>American journal of neuroradiology : AJNR, 2020-05, Vol.41 (5), p.889-897</ispartof><rights>2020 by American Journal of Neuroradiology.</rights><rights>2020 by American Journal of Neuroradiology 2020 American Journal of Neuroradiology</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c378t-44a78287baa6a2544aa704b5bd387519811de9fdf879ab1ffc00c7f24841b2dd3</citedby><cites>FETCH-LOGICAL-c378t-44a78287baa6a2544aa704b5bd387519811de9fdf879ab1ffc00c7f24841b2dd3</cites><orcidid>0000-0002-8470-4595 ; 0000-0003-0358-9810 ; 0000-0001-6491-5734 ; 0000-0002-8213-8964 ; 0000-0002-7251-2916 ; 0000-0003-1428-6348 ; 0000-0002-5986-5258</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7228188/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7228188/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,723,776,780,881,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32381544$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Yoon, H M</creatorcontrib><creatorcontrib>Jo, Y</creatorcontrib><creatorcontrib>Shim, W H</creatorcontrib><creatorcontrib>Lee, J S</creatorcontrib><creatorcontrib>Ko, T S</creatorcontrib><creatorcontrib>Koo, J H</creatorcontrib><creatorcontrib>Yum, M S</creatorcontrib><title>Disrupted Functional and Structural Connectivity in Angelman Syndrome</title><title>American journal of neuroradiology : AJNR</title><addtitle>AJNR Am J Neuroradiol</addtitle><description>This work investigated alterations in functional connectivity (FC) and associated structures in patients with Angelman syndrome (AS) by using integrated quantitative imaging analysis and connectivity measures.
We obtained 3T brain MR imaging, including resting-state functional MR imaging, diffusion tensor imaging, and 3D T1-weighted imaging from children with AS (
= 14) and age- and sex-matched controls (
= 28). The brains of patients with AS were analyzed by measuring FC, white matter microstructural analysis, cortical thickness, and brain volumes; these were compared with brains of controls.
Interregional FC analysis revealed significantly reduced intra- and interhemispheric FC, especially in the basal ganglia and thalamus, in patients with AS. Significant reductions in fractional anisotropy were found in the corpus callosum, cingulum, posterior limb of the internal capsules, and arcuate fasciculus in patients with AS. Quantitative structural analysis also showed gray matter volume loss of the basal ganglia and diffuse WM volume reduction in AS compared with the control group.
This integrated quantitative MR imaging analysis demonstrated poor functional and structural connectivity, as well as brain volume reduction, in children with AS, which may explain the motor and language dysfunction observed in this well-characterized neurobehavioral phenotype.</description><subject>Adolescent</subject><subject>Angelman Syndrome - diagnostic imaging</subject><subject>Angelman Syndrome - pathology</subject><subject>Angelman Syndrome - physiopathology</subject><subject>Brain - diagnostic imaging</subject><subject>Brain - pathology</subject><subject>Brain - physiopathology</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Diffusion Tensor Imaging - methods</subject><subject>Female</subject><subject>Functional</subject><subject>Humans</subject><subject>Image Interpretation, Computer-Assisted - methods</subject><subject>Male</subject><subject>Nerve Net - diagnostic imaging</subject><subject>Nerve Net - pathology</subject><subject>Nerve Net - physiopathology</subject><subject>Neuroimaging - methods</subject><subject>Pediatrics</subject><issn>0195-6108</issn><issn>1936-959X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpVkE1LAzEYhIMotlYv_gDZowhb87VNchFKbVUoeKiCt5DdZGvKbrYm2UL_vVtbi55eXmaYGR4ArhEcEsTovVo5PxyPMoJOQB8JMkpFJj5OQR8ikaUjBHkPXISwghBmguFz0COYcJRR2gfTRxt8u45GJ7PWFdE2TlWJcjpZRN8WsfXdO2mcM522sXGbWJeM3dJUtXLJYuu0b2pzCc5KVQVzdbgD8D6bvk2e0_nr08tkPE8LwnhMKVWMY85ypUYKd_1KMUjzLNeEswwJjpA2otQlZ0LlqCwLCAtWYsopyrHWZAAe9rnrNq-NLoyL3T659rZWfisbZeV_xdlPuWw2kmHMEeddwO0hwDdfrQlR1jYUpqqUM00bJKYdI8xFtrPe7a2Fb0LwpjzWICh33OWOu_zh3plv_g47Wn9Bk2_eE4CD</recordid><startdate>202005</startdate><enddate>202005</enddate><creator>Yoon, H M</creator><creator>Jo, Y</creator><creator>Shim, W H</creator><creator>Lee, J S</creator><creator>Ko, T S</creator><creator>Koo, J H</creator><creator>Yum, M S</creator><general>American Society of Neuroradiology</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-8470-4595</orcidid><orcidid>https://orcid.org/0000-0003-0358-9810</orcidid><orcidid>https://orcid.org/0000-0001-6491-5734</orcidid><orcidid>https://orcid.org/0000-0002-8213-8964</orcidid><orcidid>https://orcid.org/0000-0002-7251-2916</orcidid><orcidid>https://orcid.org/0000-0003-1428-6348</orcidid><orcidid>https://orcid.org/0000-0002-5986-5258</orcidid></search><sort><creationdate>202005</creationdate><title>Disrupted Functional and Structural Connectivity in Angelman Syndrome</title><author>Yoon, H M ; Jo, Y ; Shim, W H ; Lee, J S ; Ko, T S ; Koo, J H ; Yum, M S</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c378t-44a78287baa6a2544aa704b5bd387519811de9fdf879ab1ffc00c7f24841b2dd3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adolescent</topic><topic>Angelman Syndrome - diagnostic imaging</topic><topic>Angelman Syndrome - pathology</topic><topic>Angelman Syndrome - physiopathology</topic><topic>Brain - diagnostic imaging</topic><topic>Brain - pathology</topic><topic>Brain - physiopathology</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Diffusion Tensor Imaging - methods</topic><topic>Female</topic><topic>Functional</topic><topic>Humans</topic><topic>Image Interpretation, Computer-Assisted - methods</topic><topic>Male</topic><topic>Nerve Net - diagnostic imaging</topic><topic>Nerve Net - pathology</topic><topic>Nerve Net - physiopathology</topic><topic>Neuroimaging - methods</topic><topic>Pediatrics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Yoon, H M</creatorcontrib><creatorcontrib>Jo, Y</creatorcontrib><creatorcontrib>Shim, W H</creatorcontrib><creatorcontrib>Lee, J S</creatorcontrib><creatorcontrib>Ko, T S</creatorcontrib><creatorcontrib>Koo, J H</creatorcontrib><creatorcontrib>Yum, M S</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>American journal of neuroradiology : AJNR</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Yoon, H M</au><au>Jo, Y</au><au>Shim, W H</au><au>Lee, J S</au><au>Ko, T S</au><au>Koo, J H</au><au>Yum, M S</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Disrupted Functional and Structural Connectivity in Angelman Syndrome</atitle><jtitle>American journal of neuroradiology : AJNR</jtitle><addtitle>AJNR Am J Neuroradiol</addtitle><date>2020-05</date><risdate>2020</risdate><volume>41</volume><issue>5</issue><spage>889</spage><epage>897</epage><pages>889-897</pages><issn>0195-6108</issn><eissn>1936-959X</eissn><abstract>This work investigated alterations in functional connectivity (FC) and associated structures in patients with Angelman syndrome (AS) by using integrated quantitative imaging analysis and connectivity measures.
We obtained 3T brain MR imaging, including resting-state functional MR imaging, diffusion tensor imaging, and 3D T1-weighted imaging from children with AS (
= 14) and age- and sex-matched controls (
= 28). The brains of patients with AS were analyzed by measuring FC, white matter microstructural analysis, cortical thickness, and brain volumes; these were compared with brains of controls.
Interregional FC analysis revealed significantly reduced intra- and interhemispheric FC, especially in the basal ganglia and thalamus, in patients with AS. Significant reductions in fractional anisotropy were found in the corpus callosum, cingulum, posterior limb of the internal capsules, and arcuate fasciculus in patients with AS. Quantitative structural analysis also showed gray matter volume loss of the basal ganglia and diffuse WM volume reduction in AS compared with the control group.
This integrated quantitative MR imaging analysis demonstrated poor functional and structural connectivity, as well as brain volume reduction, in children with AS, which may explain the motor and language dysfunction observed in this well-characterized neurobehavioral phenotype.</abstract><cop>United States</cop><pub>American Society of Neuroradiology</pub><pmid>32381544</pmid><doi>10.3174/ajnr.A6531</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0002-8470-4595</orcidid><orcidid>https://orcid.org/0000-0003-0358-9810</orcidid><orcidid>https://orcid.org/0000-0001-6491-5734</orcidid><orcidid>https://orcid.org/0000-0002-8213-8964</orcidid><orcidid>https://orcid.org/0000-0002-7251-2916</orcidid><orcidid>https://orcid.org/0000-0003-1428-6348</orcidid><orcidid>https://orcid.org/0000-0002-5986-5258</orcidid><oa>free_for_read</oa></addata></record> |
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| source | MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central |
| subjects | Adolescent Angelman Syndrome - diagnostic imaging Angelman Syndrome - pathology Angelman Syndrome - physiopathology Brain - diagnostic imaging Brain - pathology Brain - physiopathology Child Child, Preschool Diffusion Tensor Imaging - methods Female Functional Humans Image Interpretation, Computer-Assisted - methods Male Nerve Net - diagnostic imaging Nerve Net - pathology Nerve Net - physiopathology Neuroimaging - methods Pediatrics |
| title | Disrupted Functional and Structural Connectivity in Angelman Syndrome |
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