Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study
To characterize retinal and choroidal microvascular and structural changes in patients who are gene positive for mutant huntingtin protein (mHtt) with symptoms of Huntington's Disease (HD). This study is a cross-sectional comparison of patients who are gene positive for mHtt and exhibit symptom...
Gespeichert in:
| Veröffentlicht in: | PloS one 2024-01, Vol.19 (1), p.e0296742 |
|---|---|
| Hauptverfasser: | , , , , , , , , , , , , , |
| Format: | Artikel |
| Sprache: | eng |
| Schlagworte: | |
| Online-Zugang: | Volltext |
| Tags: |
Tag hinzufügen
Keine Tags, Fügen Sie den ersten Tag hinzu!
|
| container_end_page | |
|---|---|
| container_issue | 1 |
| container_start_page | e0296742 |
| container_title | PloS one |
| container_volume | 19 |
| creator | Joseph, Suzanna Robbins, Cason B Haystead, Alice Hemesath, Angela Allen, Ariana Kundu, Anita Ma, Justin P Scott, Burton L Moore, Kathryn P L Agrawal, Rupesh Gunasan, Vithiya Stinnett, Sandra S Grewal, Dilraj S Fekrat, Sharon |
| description | To characterize retinal and choroidal microvascular and structural changes in patients who are gene positive for mutant huntingtin protein (mHtt) with symptoms of Huntington's Disease (HD).
This study is a cross-sectional comparison of patients who are gene positive for mHtt and exhibit symptoms of HD, either motor manifest or prodromal (HD group), and cognitively normal individuals without a family history of HD (control group). HD patients were diagnosed by Duke movement disorder neurologists based on the Unified Huntington's Disease Rating Scale (UHDRS). Fovea and optic nerve centered OCT and OCTA images were captured using Zeiss Cirrus HD-5000 with AngioPlex. Outcome metrics included central subfield thickness (CST), peripapillary retinal nerve fiber layer (pRNFL) thickness, ganglion cell-inner plexiform layer (GCIPL) thickness, and choroidal vascularity index (CVI) on OCT, and foveal avascular zone (FAZ) area, vessel density (VD), perfusion density (PD), capillary perfusion density (CPD), and capillary flux index (CFI) on OCTA. Generalized estimating equation (GEE) models were used to account for inter-eye correlation.
Forty-four eyes of 23 patients in the HD group and 77 eyes of 39 patients in the control group were analyzed. Average GCIPL thickness and FAZ area were decreased in the HD group compared to controls (p = 0.001, p < 0.001). No other imaging metrics were significantly different between groups.
Patients in the HD group had decreased GCIPL thickness and smaller FAZ area, highlighting the potential use of retinal biomarkers in detecting neurodegenerative changes in HD. |
| doi_str_mv | 10.1371/journal.pone.0296742 |
| format | Article |
| fullrecord | <record><control><sourceid>gale_plos_</sourceid><recordid>TN_cdi_plos_journals_3069265620</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><galeid>A780936844</galeid><doaj_id>oai_doaj_org_article_557380510b3d4b17ad2d2c52bd45fb4a</doaj_id><sourcerecordid>A780936844</sourcerecordid><originalsourceid>FETCH-LOGICAL-c642t-e1f6708b790c462a32485954336f9e980e023b00e4f6b4bb2727e774a9f6499e3</originalsourceid><addsrcrecordid>eNqNk9tu1DAQhiMEomXhDRBYQuJwsYtjO07MDarKoZUqVeJ0azn2ZNdV1t7azkJ5N94NZ3dbdVEvUC6Ssb_5_3g8UxRPSzwraV2-vfBDcKqfrbyDGSaC14zcKw5LQcmUE0zv3_o-KB7FeIFxRRvOHxYHtCGNEKU4LP4cL1RQOkGwv62bI2O7DgI4DRFZhwIkm02QcgbphQ_emhwtrQ5-raIeepWGAJvtmMKgN1HOs87YtTWD6iP6adMCnQwuK82Td68i-mAjqAhI--VKBTAoebQA1afFVV5zKfg-vkNHKLvEOI2gk_XjX6xyvBqjNWS7wVw9Lh502QKe7N6T4vunj9-OT6Zn559Pj4_OppozkqZQdrzGTVsLrBknihLWVKJilPJOgGgwYEJbjIF1vGVtS2pSQ10zJTrOhAA6KZ5vdVe9j3JX-Sgp5oLwaqzwpDjdEsarC7kKdqnClfTKys2CD3OpQrK6B1lVNW1wVeKWGtaWtTLEEF2R1rCqa5nKWu93bkO7BKMhV0T1e6L7O84u5NyvZYkbwkXFs8LrnULwlwPEJJc2auh75cAPURJBcFWXJa0y-uIf9O7j7ai5yiewrvPZWI-i8qhusKC8YSxTszuo_BjILZP7tLN5fS_hzV7CePnwK83VEKM8_frl_9nzH_vsy1vstrWi74exj-I-yLbgptUCdDdVLrEcx-y6GnIcM7kbs5z27PYN3SRdzxX9Cxe9JwA</addsrcrecordid><sourcetype>Open Website</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>3069265620</pqid></control><display><type>article</type><title>Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study</title><source>Public Library of Science (PLoS) Journals Open Access</source><source>MEDLINE</source><source>DOAJ Directory of Open Access Journals</source><source>EZB-FREE-00999 freely available EZB journals</source><source>PubMed Central</source><source>Free Full-Text Journals in Chemistry</source><creator>Joseph, Suzanna ; Robbins, Cason B ; Haystead, Alice ; Hemesath, Angela ; Allen, Ariana ; Kundu, Anita ; Ma, Justin P ; Scott, Burton L ; Moore, Kathryn P L ; Agrawal, Rupesh ; Gunasan, Vithiya ; Stinnett, Sandra S ; Grewal, Dilraj S ; Fekrat, Sharon</creator><contributor>Cicero, Calogero Edoardo</contributor><creatorcontrib>Joseph, Suzanna ; Robbins, Cason B ; Haystead, Alice ; Hemesath, Angela ; Allen, Ariana ; Kundu, Anita ; Ma, Justin P ; Scott, Burton L ; Moore, Kathryn P L ; Agrawal, Rupesh ; Gunasan, Vithiya ; Stinnett, Sandra S ; Grewal, Dilraj S ; Fekrat, Sharon ; Cicero, Calogero Edoardo</creatorcontrib><description>To characterize retinal and choroidal microvascular and structural changes in patients who are gene positive for mutant huntingtin protein (mHtt) with symptoms of Huntington's Disease (HD).
This study is a cross-sectional comparison of patients who are gene positive for mHtt and exhibit symptoms of HD, either motor manifest or prodromal (HD group), and cognitively normal individuals without a family history of HD (control group). HD patients were diagnosed by Duke movement disorder neurologists based on the Unified Huntington's Disease Rating Scale (UHDRS). Fovea and optic nerve centered OCT and OCTA images were captured using Zeiss Cirrus HD-5000 with AngioPlex. Outcome metrics included central subfield thickness (CST), peripapillary retinal nerve fiber layer (pRNFL) thickness, ganglion cell-inner plexiform layer (GCIPL) thickness, and choroidal vascularity index (CVI) on OCT, and foveal avascular zone (FAZ) area, vessel density (VD), perfusion density (PD), capillary perfusion density (CPD), and capillary flux index (CFI) on OCTA. Generalized estimating equation (GEE) models were used to account for inter-eye correlation.
Forty-four eyes of 23 patients in the HD group and 77 eyes of 39 patients in the control group were analyzed. Average GCIPL thickness and FAZ area were decreased in the HD group compared to controls (p = 0.001, p < 0.001). No other imaging metrics were significantly different between groups.
Patients in the HD group had decreased GCIPL thickness and smaller FAZ area, highlighting the potential use of retinal biomarkers in detecting neurodegenerative changes in HD.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0296742</identifier><identifier>PMID: 38289919</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Analysis ; Biological markers ; Biology and Life Sciences ; Biomarkers ; Care and treatment ; Cognition & reasoning ; Cross-Sectional Studies ; Data analysis ; Data collection ; Density ; Development and progression ; Diagnosis ; Disease control ; Disease prevention ; Eye ; Family medical history ; Fluorescein Angiography - methods ; Fovea ; Genetics ; High-definition television ; Humans ; Huntingtin ; Huntington Disease - diagnostic imaging ; Huntington's chorea ; Huntington's disease ; Huntingtons disease ; Medical records ; Medical research ; Medicine and Health Sciences ; Medicine, Experimental ; Microvasculature ; Microvessels - diagnostic imaging ; Movement disorders ; Nerves ; Neurological disorders ; Ocular manifestations of general diseases ; Optic nerve ; Optics ; Patient outcomes ; Perfusion ; Prospective Studies ; Research and Analysis Methods ; Retina ; Retinal Ganglion Cells ; Retinal Vessels - diagnostic imaging ; Sexually transmitted diseases ; Signs and symptoms ; Social Sciences ; Software ; STD ; Thickness ; Tomography, Optical Coherence - methods ; Visual acuity</subject><ispartof>PloS one, 2024-01, Vol.19 (1), p.e0296742</ispartof><rights>Copyright: © 2024 Joseph et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.</rights><rights>COPYRIGHT 2024 Public Library of Science</rights><rights>2024 Joseph et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2024 Joseph et al 2024 Joseph et al</rights><rights>2024 Joseph et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c642t-e1f6708b790c462a32485954336f9e980e023b00e4f6b4bb2727e774a9f6499e3</cites><orcidid>0000-0002-4495-7221 ; 0000-0003-0003-158X ; 0000-0001-7909-510X ; 0000-0003-4403-5996</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/PMC10826956/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC10826956/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,860,881,2096,2915,23847,27903,27904,53769,53771,79346,79347</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/38289919$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><contributor>Cicero, Calogero Edoardo</contributor><creatorcontrib>Joseph, Suzanna</creatorcontrib><creatorcontrib>Robbins, Cason B</creatorcontrib><creatorcontrib>Haystead, Alice</creatorcontrib><creatorcontrib>Hemesath, Angela</creatorcontrib><creatorcontrib>Allen, Ariana</creatorcontrib><creatorcontrib>Kundu, Anita</creatorcontrib><creatorcontrib>Ma, Justin P</creatorcontrib><creatorcontrib>Scott, Burton L</creatorcontrib><creatorcontrib>Moore, Kathryn P L</creatorcontrib><creatorcontrib>Agrawal, Rupesh</creatorcontrib><creatorcontrib>Gunasan, Vithiya</creatorcontrib><creatorcontrib>Stinnett, Sandra S</creatorcontrib><creatorcontrib>Grewal, Dilraj S</creatorcontrib><creatorcontrib>Fekrat, Sharon</creatorcontrib><title>Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>To characterize retinal and choroidal microvascular and structural changes in patients who are gene positive for mutant huntingtin protein (mHtt) with symptoms of Huntington's Disease (HD).
This study is a cross-sectional comparison of patients who are gene positive for mHtt and exhibit symptoms of HD, either motor manifest or prodromal (HD group), and cognitively normal individuals without a family history of HD (control group). HD patients were diagnosed by Duke movement disorder neurologists based on the Unified Huntington's Disease Rating Scale (UHDRS). Fovea and optic nerve centered OCT and OCTA images were captured using Zeiss Cirrus HD-5000 with AngioPlex. Outcome metrics included central subfield thickness (CST), peripapillary retinal nerve fiber layer (pRNFL) thickness, ganglion cell-inner plexiform layer (GCIPL) thickness, and choroidal vascularity index (CVI) on OCT, and foveal avascular zone (FAZ) area, vessel density (VD), perfusion density (PD), capillary perfusion density (CPD), and capillary flux index (CFI) on OCTA. Generalized estimating equation (GEE) models were used to account for inter-eye correlation.
Forty-four eyes of 23 patients in the HD group and 77 eyes of 39 patients in the control group were analyzed. Average GCIPL thickness and FAZ area were decreased in the HD group compared to controls (p = 0.001, p < 0.001). No other imaging metrics were significantly different between groups.
Patients in the HD group had decreased GCIPL thickness and smaller FAZ area, highlighting the potential use of retinal biomarkers in detecting neurodegenerative changes in HD.</description><subject>Analysis</subject><subject>Biological markers</subject><subject>Biology and Life Sciences</subject><subject>Biomarkers</subject><subject>Care and treatment</subject><subject>Cognition & reasoning</subject><subject>Cross-Sectional Studies</subject><subject>Data analysis</subject><subject>Data collection</subject><subject>Density</subject><subject>Development and progression</subject><subject>Diagnosis</subject><subject>Disease control</subject><subject>Disease prevention</subject><subject>Eye</subject><subject>Family medical history</subject><subject>Fluorescein Angiography - methods</subject><subject>Fovea</subject><subject>Genetics</subject><subject>High-definition television</subject><subject>Humans</subject><subject>Huntingtin</subject><subject>Huntington Disease - diagnostic imaging</subject><subject>Huntington's chorea</subject><subject>Huntington's disease</subject><subject>Huntingtons disease</subject><subject>Medical records</subject><subject>Medical research</subject><subject>Medicine and Health Sciences</subject><subject>Medicine, Experimental</subject><subject>Microvasculature</subject><subject>Microvessels - diagnostic imaging</subject><subject>Movement disorders</subject><subject>Nerves</subject><subject>Neurological disorders</subject><subject>Ocular manifestations of general diseases</subject><subject>Optic nerve</subject><subject>Optics</subject><subject>Patient outcomes</subject><subject>Perfusion</subject><subject>Prospective Studies</subject><subject>Research and Analysis Methods</subject><subject>Retina</subject><subject>Retinal Ganglion Cells</subject><subject>Retinal Vessels - diagnostic imaging</subject><subject>Sexually transmitted diseases</subject><subject>Signs and symptoms</subject><subject>Social Sciences</subject><subject>Software</subject><subject>STD</subject><subject>Thickness</subject><subject>Tomography, Optical Coherence - methods</subject><subject>Visual acuity</subject><issn>1932-6203</issn><issn>1932-6203</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><sourceid>DOA</sourceid><recordid>eNqNk9tu1DAQhiMEomXhDRBYQuJwsYtjO07MDarKoZUqVeJ0azn2ZNdV1t7azkJ5N94NZ3dbdVEvUC6Ssb_5_3g8UxRPSzwraV2-vfBDcKqfrbyDGSaC14zcKw5LQcmUE0zv3_o-KB7FeIFxRRvOHxYHtCGNEKU4LP4cL1RQOkGwv62bI2O7DgI4DRFZhwIkm02QcgbphQ_emhwtrQ5-raIeepWGAJvtmMKgN1HOs87YtTWD6iP6adMCnQwuK82Td68i-mAjqAhI--VKBTAoebQA1afFVV5zKfg-vkNHKLvEOI2gk_XjX6xyvBqjNWS7wVw9Lh502QKe7N6T4vunj9-OT6Zn559Pj4_OppozkqZQdrzGTVsLrBknihLWVKJilPJOgGgwYEJbjIF1vGVtS2pSQ10zJTrOhAA6KZ5vdVe9j3JX-Sgp5oLwaqzwpDjdEsarC7kKdqnClfTKys2CD3OpQrK6B1lVNW1wVeKWGtaWtTLEEF2R1rCqa5nKWu93bkO7BKMhV0T1e6L7O84u5NyvZYkbwkXFs8LrnULwlwPEJJc2auh75cAPURJBcFWXJa0y-uIf9O7j7ai5yiewrvPZWI-i8qhusKC8YSxTszuo_BjILZP7tLN5fS_hzV7CePnwK83VEKM8_frl_9nzH_vsy1vstrWi74exj-I-yLbgptUCdDdVLrEcx-y6GnIcM7kbs5z27PYN3SRdzxX9Cxe9JwA</recordid><startdate>20240130</startdate><enddate>20240130</enddate><creator>Joseph, Suzanna</creator><creator>Robbins, Cason B</creator><creator>Haystead, Alice</creator><creator>Hemesath, Angela</creator><creator>Allen, Ariana</creator><creator>Kundu, Anita</creator><creator>Ma, Justin P</creator><creator>Scott, Burton L</creator><creator>Moore, Kathryn P L</creator><creator>Agrawal, Rupesh</creator><creator>Gunasan, Vithiya</creator><creator>Stinnett, Sandra S</creator><creator>Grewal, Dilraj S</creator><creator>Fekrat, Sharon</creator><general>Public Library of Science</general><general>Public Library of Science (PLoS)</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>IOV</scope><scope>ISR</scope><scope>3V.</scope><scope>7QG</scope><scope>7QL</scope><scope>7QO</scope><scope>7RV</scope><scope>7SN</scope><scope>7SS</scope><scope>7T5</scope><scope>7TG</scope><scope>7TM</scope><scope>7U9</scope><scope>7X2</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FD</scope><scope>8FE</scope><scope>8FG</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABJCF</scope><scope>ABUWG</scope><scope>AEUYN</scope><scope>AFKRA</scope><scope>ARAPS</scope><scope>ATCPS</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BGLVJ</scope><scope>BHPHI</scope><scope>C1K</scope><scope>CCPQU</scope><scope>D1I</scope><scope>DWQXO</scope><scope>FR3</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>H94</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>KB.</scope><scope>KB0</scope><scope>KL.</scope><scope>L6V</scope><scope>LK8</scope><scope>M0K</scope><scope>M0S</scope><scope>M1P</scope><scope>M7N</scope><scope>M7P</scope><scope>M7S</scope><scope>NAPCQ</scope><scope>P5Z</scope><scope>P62</scope><scope>P64</scope><scope>PATMY</scope><scope>PDBOC</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>PTHSS</scope><scope>PYCSY</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><scope>DOA</scope><orcidid>https://orcid.org/0000-0002-4495-7221</orcidid><orcidid>https://orcid.org/0000-0003-0003-158X</orcidid><orcidid>https://orcid.org/0000-0001-7909-510X</orcidid><orcidid>https://orcid.org/0000-0003-4403-5996</orcidid></search><sort><creationdate>20240130</creationdate><title>Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study</title><author>Joseph, Suzanna ; Robbins, Cason B ; Haystead, Alice ; Hemesath, Angela ; Allen, Ariana ; Kundu, Anita ; Ma, Justin P ; Scott, Burton L ; Moore, Kathryn P L ; Agrawal, Rupesh ; Gunasan, Vithiya ; Stinnett, Sandra S ; Grewal, Dilraj S ; Fekrat, Sharon</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c642t-e1f6708b790c462a32485954336f9e980e023b00e4f6b4bb2727e774a9f6499e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Analysis</topic><topic>Biological markers</topic><topic>Biology and Life Sciences</topic><topic>Biomarkers</topic><topic>Care and treatment</topic><topic>Cognition & reasoning</topic><topic>Cross-Sectional Studies</topic><topic>Data analysis</topic><topic>Data collection</topic><topic>Density</topic><topic>Development and progression</topic><topic>Diagnosis</topic><topic>Disease control</topic><topic>Disease prevention</topic><topic>Eye</topic><topic>Family medical history</topic><topic>Fluorescein Angiography - methods</topic><topic>Fovea</topic><topic>Genetics</topic><topic>High-definition television</topic><topic>Humans</topic><topic>Huntingtin</topic><topic>Huntington Disease - diagnostic imaging</topic><topic>Huntington's chorea</topic><topic>Huntington's disease</topic><topic>Huntingtons disease</topic><topic>Medical records</topic><topic>Medical research</topic><topic>Medicine and Health Sciences</topic><topic>Medicine, Experimental</topic><topic>Microvasculature</topic><topic>Microvessels - diagnostic imaging</topic><topic>Movement disorders</topic><topic>Nerves</topic><topic>Neurological disorders</topic><topic>Ocular manifestations of general diseases</topic><topic>Optic nerve</topic><topic>Optics</topic><topic>Patient outcomes</topic><topic>Perfusion</topic><topic>Prospective Studies</topic><topic>Research and Analysis Methods</topic><topic>Retina</topic><topic>Retinal Ganglion Cells</topic><topic>Retinal Vessels - diagnostic imaging</topic><topic>Sexually transmitted diseases</topic><topic>Signs and symptoms</topic><topic>Social Sciences</topic><topic>Software</topic><topic>STD</topic><topic>Thickness</topic><topic>Tomography, Optical Coherence - methods</topic><topic>Visual acuity</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Joseph, Suzanna</creatorcontrib><creatorcontrib>Robbins, Cason B</creatorcontrib><creatorcontrib>Haystead, Alice</creatorcontrib><creatorcontrib>Hemesath, Angela</creatorcontrib><creatorcontrib>Allen, Ariana</creatorcontrib><creatorcontrib>Kundu, Anita</creatorcontrib><creatorcontrib>Ma, Justin P</creatorcontrib><creatorcontrib>Scott, Burton L</creatorcontrib><creatorcontrib>Moore, Kathryn P L</creatorcontrib><creatorcontrib>Agrawal, Rupesh</creatorcontrib><creatorcontrib>Gunasan, Vithiya</creatorcontrib><creatorcontrib>Stinnett, Sandra S</creatorcontrib><creatorcontrib>Grewal, Dilraj S</creatorcontrib><creatorcontrib>Fekrat, Sharon</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Gale In Context: Opposing Viewpoints</collection><collection>Gale In Context: Science</collection><collection>ProQuest Central (Corporate)</collection><collection>Animal Behavior Abstracts</collection><collection>Bacteriology Abstracts (Microbiology B)</collection><collection>Biotechnology Research Abstracts</collection><collection>Nursing & Allied Health Database</collection><collection>Ecology Abstracts</collection><collection>Entomology Abstracts (Full archive)</collection><collection>Immunology Abstracts</collection><collection>Meteorological & Geoastrophysical Abstracts</collection><collection>Nucleic Acids Abstracts</collection><collection>Virology and AIDS Abstracts</collection><collection>Agricultural Science Collection</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Public Health Database</collection><collection>Technology Research Database</collection><collection>ProQuest SciTech Collection</collection><collection>ProQuest Technology Collection</collection><collection>ProQuest Natural Science Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>Materials Science & Engineering Collection</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest One Sustainability</collection><collection>ProQuest Central UK/Ireland</collection><collection>Advanced Technologies & Aerospace Collection</collection><collection>Agricultural & Environmental Science Collection</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>ProQuest Central</collection><collection>Technology Collection</collection><collection>Natural Science Collection</collection><collection>Environmental Sciences and Pollution Management</collection><collection>ProQuest One Community College</collection><collection>ProQuest Materials Science Collection</collection><collection>ProQuest Central Korea</collection><collection>Engineering Research Database</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Materials Science Database</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Meteorological & Geoastrophysical Abstracts - Academic</collection><collection>ProQuest Engineering Collection</collection><collection>ProQuest Biological Science Collection</collection><collection>Agricultural Science Database</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Algology Mycology and Protozoology Abstracts (Microbiology C)</collection><collection>Biological Science Database</collection><collection>Engineering Database</collection><collection>Nursing & Allied Health Premium</collection><collection>Advanced Technologies & Aerospace Database</collection><collection>ProQuest Advanced Technologies & Aerospace Collection</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Environmental Science Database</collection><collection>Materials Science Collection</collection><collection>Publicly Available Content Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>Engineering Collection</collection><collection>Environmental Science Collection</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>PloS one</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Joseph, Suzanna</au><au>Robbins, Cason B</au><au>Haystead, Alice</au><au>Hemesath, Angela</au><au>Allen, Ariana</au><au>Kundu, Anita</au><au>Ma, Justin P</au><au>Scott, Burton L</au><au>Moore, Kathryn P L</au><au>Agrawal, Rupesh</au><au>Gunasan, Vithiya</au><au>Stinnett, Sandra S</au><au>Grewal, Dilraj S</au><au>Fekrat, Sharon</au><au>Cicero, Calogero Edoardo</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2024-01-30</date><risdate>2024</risdate><volume>19</volume><issue>1</issue><spage>e0296742</spage><pages>e0296742-</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>To characterize retinal and choroidal microvascular and structural changes in patients who are gene positive for mutant huntingtin protein (mHtt) with symptoms of Huntington's Disease (HD).
This study is a cross-sectional comparison of patients who are gene positive for mHtt and exhibit symptoms of HD, either motor manifest or prodromal (HD group), and cognitively normal individuals without a family history of HD (control group). HD patients were diagnosed by Duke movement disorder neurologists based on the Unified Huntington's Disease Rating Scale (UHDRS). Fovea and optic nerve centered OCT and OCTA images were captured using Zeiss Cirrus HD-5000 with AngioPlex. Outcome metrics included central subfield thickness (CST), peripapillary retinal nerve fiber layer (pRNFL) thickness, ganglion cell-inner plexiform layer (GCIPL) thickness, and choroidal vascularity index (CVI) on OCT, and foveal avascular zone (FAZ) area, vessel density (VD), perfusion density (PD), capillary perfusion density (CPD), and capillary flux index (CFI) on OCTA. Generalized estimating equation (GEE) models were used to account for inter-eye correlation.
Forty-four eyes of 23 patients in the HD group and 77 eyes of 39 patients in the control group were analyzed. Average GCIPL thickness and FAZ area were decreased in the HD group compared to controls (p = 0.001, p < 0.001). No other imaging metrics were significantly different between groups.
Patients in the HD group had decreased GCIPL thickness and smaller FAZ area, highlighting the potential use of retinal biomarkers in detecting neurodegenerative changes in HD.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>38289919</pmid><doi>10.1371/journal.pone.0296742</doi><tpages>e0296742</tpages><orcidid>https://orcid.org/0000-0002-4495-7221</orcidid><orcidid>https://orcid.org/0000-0003-0003-158X</orcidid><orcidid>https://orcid.org/0000-0001-7909-510X</orcidid><orcidid>https://orcid.org/0000-0003-4403-5996</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 1932-6203 |
| ispartof | PloS one, 2024-01, Vol.19 (1), p.e0296742 |
| issn | 1932-6203 1932-6203 |
| language | eng |
| recordid | cdi_plos_journals_3069265620 |
| source | Public Library of Science (PLoS) Journals Open Access; MEDLINE; DOAJ Directory of Open Access Journals; EZB-FREE-00999 freely available EZB journals; PubMed Central; Free Full-Text Journals in Chemistry |
| subjects | Analysis Biological markers Biology and Life Sciences Biomarkers Care and treatment Cognition & reasoning Cross-Sectional Studies Data analysis Data collection Density Development and progression Diagnosis Disease control Disease prevention Eye Family medical history Fluorescein Angiography - methods Fovea Genetics High-definition television Humans Huntingtin Huntington Disease - diagnostic imaging Huntington's chorea Huntington's disease Huntingtons disease Medical records Medical research Medicine and Health Sciences Medicine, Experimental Microvasculature Microvessels - diagnostic imaging Movement disorders Nerves Neurological disorders Ocular manifestations of general diseases Optic nerve Optics Patient outcomes Perfusion Prospective Studies Research and Analysis Methods Retina Retinal Ganglion Cells Retinal Vessels - diagnostic imaging Sexually transmitted diseases Signs and symptoms Social Sciences Software STD Thickness Tomography, Optical Coherence - methods Visual acuity |
| title | Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls: A cross-sectional prospective study |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2025-01-27T11%3A04%3A21IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-gale_plos_&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Characterizing%20differences%20in%20retinal%20and%20choroidal%20microvasculature%20and%20structure%20in%20individuals%20with%20Huntington's%20Disease%20compared%20to%20healthy%20controls:%20A%20cross-sectional%20prospective%20study&rft.jtitle=PloS%20one&rft.au=Joseph,%20Suzanna&rft.date=2024-01-30&rft.volume=19&rft.issue=1&rft.spage=e0296742&rft.pages=e0296742-&rft.issn=1932-6203&rft.eissn=1932-6203&rft_id=info:doi/10.1371/journal.pone.0296742&rft_dat=%3Cgale_plos_%3EA780936844%3C/gale_plos_%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=3069265620&rft_id=info:pmid/38289919&rft_galeid=A780936844&rft_doaj_id=oai_doaj_org_article_557380510b3d4b17ad2d2c52bd45fb4a&rfr_iscdi=true |