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dc.rights.licenseAttribution 4.0 International*
dc.contributor.authorRomero-Bascones, David
dc.contributor.authorAyala, Unai
dc.contributor.otherMurueta Goyena, Ane
dc.contributor.otherdel Pino, Rocío
dc.contributor.otherAcera, Marian
dc.contributor.otherTeijeira‑Portas, Sara
dc.contributor.otherFernández‑Valle, Tamara
dc.contributor.otherTijero, Beatriz
dc.contributor.otherGabilondo, Iñigo
dc.contributor.otherGómez Esteban, Juan Carlos
dc.date.accessioned2023-09-05T15:55:29Z
dc.date.available2023-09-05T15:55:29Z
dc.date.issued2023
dc.identifier.issn0340-5354en
dc.identifier.otherhttps://katalogoa.mondragon.edu/janium-bin/janium_login_opac.pl?find&ficha_no=172728en
dc.identifier.urihttps://hdl.handle.net/20.500.11984/6106
dc.description.abstractBackground Cognitive decline has been reported in premanifest and manifest Huntington’s disease but reliable biomarkers are lacking. Inner retinal layer thickness seems to be a good biomarker of cognition in other neurodegenerative diseases. Objective To explore the relationship between optical coherence tomography-derived metrics and global cognition in Huntington’s Disease. Methods Thirty-six patients with Huntington’s disease (16 premanifest and 20 manifest) and 36 controls matched by age, sex, smoking status, and hypertension status underwent macular volumetric and peripapillary optical coherence tomography scans. Disease duration, motor status, global cognition and CAG repeats were recorded in patients. Group differences in imaging parameters and their association with clinical outcomes were analyzed using linear mixed-effect models. Results Premanifest and manifest Huntington’s disease patients presented thinner retinal external limiting membrane-Bruch’s membrane complex, and manifest patients had thinner temporal peripapillary retinal nerve fiber layer compared to controls. In manifest Huntington’s disease, macular thickness was significantly associated with MoCA scores, inner nuclear layer showing the largest regression coefficients. This relationship was consistent after adjusting for age, sex, and education and p-value correction with False Discovery Rate. None of the retinal variables were related to Unified Huntington’s Disease Rating Scale score, disease duration, or disease burden. Premanifest patients did not show a significant association between OCT-derived parameters and clinical outcomes in corrected models. Conclusions In line with other neurodegenerative diseases, OCT is a potential biomarker of cognitive status in manifest HD. Future prospective studies are needed to evaluate OCT as a potential surrogate marker of cognitive decline in HD.en
dc.description.sponsorshipGobierno Vascoes
dc.language.isoengen
dc.publisherSpringer Natureen
dc.rights© Tha Author(s) 2023en
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/*
dc.subjectHuntington's diseaseen
dc.subjectretinaen
dc.subjectoptical coherence tomographyen
dc.subjectcognitionen
dc.titleRetinal thickness as a biomarker of cognitive impairment in manifest Huntington’s diseaseen
dcterms.accessRightshttp://purl.org/coar/access_right/c_abf2en
dcterms.sourceJournal of Neurologyen
local.contributor.groupTeoría de la señal y comunicacioneses
local.description.peerreviewedtrueen
local.description.publicationfirstpage3821en
local.description.publicationlastpage3829en
local.identifier.doihttps://doi.org/10.1007/s00415-023-11720-3en
local.relation.projectIDinfo:eu-repo/grantAgreement/GV/Convocatoria 2019. Línea 1 Promoción de la actividad investigadora sanitaria/2019111004/CAPV/Identificación de predictores clínicos para la detección precoz de trastornos neuropsicólogos y neuropsiquiátricos severos en portadores asintomáticos y sintomáticos de enfermedad de Huntington/en
local.relation.projectIDinfo:eu-repo/grantAgreement/GV//BIO17-ND-009/CAPV/Identificación de predictores clínicos para la detección precoz de trastornos neuropsicólogos y neuropsiquiátricos severos en portadores asintomáticos y sintomáticos de enfermedad de Huntington/en
local.contributor.otherinstitutionhttps://ror.org/0061s4v88es
local.contributor.otherinstitutionUPV/EHUes
local.contributor.otherinstitutionhttps://ror.org/03nzegx43es
local.contributor.otherinstitutionhttps://ror.org/01cc3fy72es
local.source.detailsVol. 270en
oaire.format.mimetypeapplication/pdfen
oaire.file$DSPACE\assetstoreen
oaire.resourceTypehttp://purl.org/coar/resource_type/c_6501en
oaire.versionhttp://purl.org/coar/version/c_970fb48d4fbd8a85en


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