Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls : A cross-sectional prospective study
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..
OBJECTIVE: 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).
METHODS: 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.
RESULTS: 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.
CONCLUSIONS: 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.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2024 |
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| Erschienen: |
2024 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:19 |
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| Enthalten in: |
PloS one - 19(2024), 1 vom: 01., Seite e0296742 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Joseph, Suzanna [VerfasserIn] |
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| Anmerkungen: |
Date Completed 01.02.2024 Date Revised 01.02.2024 published: Electronic-eCollection Citation Status MEDLINE |
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| doi: |
10.1371/journal.pone.0296742 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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NLM367802570 |
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| 100 | 1 | |a Joseph, Suzanna |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Characterizing differences in retinal and choroidal microvasculature and structure in individuals with Huntington's Disease compared to healthy controls |b A cross-sectional prospective study |
| 264 | 1 | |c 2024 | |
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| 500 | |a Date Revised 01.02.2024 | ||
| 500 | |a published: Electronic-eCollection | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a 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. | ||
| 520 | |a OBJECTIVE: 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) | ||
| 520 | |a METHODS: 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 | ||
| 520 | |a RESULTS: 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 | ||
| 520 | |a CONCLUSIONS: 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 | ||
| 650 | 4 | |a Journal Article | |
| 700 | 1 | |a Robbins, Cason B |e verfasserin |4 aut | |
| 700 | 1 | |a Haystead, Alice |e verfasserin |4 aut | |
| 700 | 1 | |a Hemesath, Angela |e verfasserin |4 aut | |
| 700 | 1 | |a Allen, Ariana |e verfasserin |4 aut | |
| 700 | 1 | |a Kundu, Anita |e verfasserin |4 aut | |
| 700 | 1 | |a Ma, Justin P |e verfasserin |4 aut | |
| 700 | 1 | |a Scott, Burton L |e verfasserin |4 aut | |
| 700 | 1 | |a Moore, Kathryn P L |e verfasserin |4 aut | |
| 700 | 1 | |a Agrawal, Rupesh |e verfasserin |4 aut | |
| 700 | 1 | |a Gunasan, Vithiya |e verfasserin |4 aut | |
| 700 | 1 | |a Stinnett, Sandra S |e verfasserin |4 aut | |
| 700 | 1 | |a Grewal, Dilraj S |e verfasserin |4 aut | |
| 700 | 1 | |a Fekrat, Sharon |e verfasserin |4 aut | |
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