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Diagnostic value of cerebrospinal fluid alpha-synuclein seed quantification in synucleinopathies

dc.rights.licenseopenen_US
dc.contributor.authorPOGGIOLINI, Ilaria
dc.contributor.authorGUPTA, Vandana
dc.contributor.authorLAWTON, Michael
dc.contributor.authorLEE, Seoyun
dc.contributor.authorEL-TURABI, Aadil
dc.contributor.authorQUEREJETA-COMA, Agustin
dc.contributor.authorTRENKWALDER, Claudia
dc.contributor.authorSIXEL-DORING, Friederike
hal.structure.identifierInstitut des Maladies Neurodégénératives [Bordeaux] [IMN]
dc.contributor.authorSAMIER FOUBERT, Alexandra
dc.contributor.authorPAVY-LE TRAON, Anne
dc.contributor.authorPLAZZI, Giuseppe
dc.contributor.authorBISCARINI, Francesco
dc.contributor.authorMONTPLAISIR, Jacques
dc.contributor.authorGAGNON, Jean Francois
dc.contributor.authorPOSTUMA, Ronald B.
dc.contributor.authorANTELMI, Elena
hal.structure.identifierInstitut des Maladies Neurodégénératives [Bordeaux] [IMN]
dc.contributor.authorMEISSNER, Wassilios
IDREF: 113664761
dc.contributor.authorMOLLENHAUER, Brit
dc.contributor.authorBEN-SHLOMO, Yoav
dc.contributor.authorHU, Michele T.
dc.contributor.authorPARKKINEN, Laura
dc.date.accessioned2022-01-19T08:35:38Z
dc.date.available2022-01-19T08:35:38Z
dc.date.issued2021-12-11
dc.identifier.issn1460-2156 (Electronic) 0006-8950 (Linking)en_US
dc.identifier.urihttps://oskar-bordeaux.fr/handle/20.500.12278/124432
dc.description.abstractEnSeveral studies have confirmed α-synuclein real-time quaking-induced conversion (αSyn-RT-QuIC) assay to have high sensitivity and specificity for Parkinson's disease. However, whether the assay can be used as a robust, quantitative measure to monitor disease progression, stratify different synucleinopathies and predict disease conversion in patients with idiopathic REM sleep behaviour disorder remains undetermined. The aim of this study was to assess the diagnostic value of CSF aSyn-RT-QuIC quantitative parameters in regard to disease progression, stratification, and conversion in synucleinopathies. We performed αSyn-RT-QuIC in the CSF samples from 74 Parkinson's disease, 24 multiple system atrophy and 45 idiopathic REM sleep behaviour disorder patients alongside 55 healthy controls, analysing quantitative assay parameters in relation to clinical data. αSyn-RT-QuIC showed 89% sensitivity and 96% specificity for Parkinson's disease. There was no correlation between RT-QuIC quantitative parameters and Parkinson's disease clinical scores (e.g. UPDRS motor) but RT-QuIC positivity and some quantitative parameters (e.g. Vmax) differed across the different phenotype clusters. RT-QuIC parameters also added value alongside standard clinical data in diagnosing Parkinson's disease. The sensitivity in multiple system atrophy was 75%, and CSF samples showed longer T50 and lower Vmax compared to Parkinson's disease. All RT-QuIC parameters correlated with worse clinical progression of multiple system atrophy (e.g. change in UMSARS). The overall sensitivity in idiopathic REM sleep behaviour disorder was 64%. In three of the four longitudinally followed idiopathic REM sleep behaviour disorder cohorts, we found around 90% sensitivity, but in one sample (DeNoPa) diagnosing idiopathic REM sleep behaviour disorder earlier from the community cases, this was much lower 39%. During follow-up, 14 of 45 (31%) idiopathic REM sleep behaviour disorder patients converted to synucleinopathy with 9/14 (64%) of convertors showing baseline RT-QuIC positivity. In summary, our results showed that αSyn-RT-QuIC adds value in diagnosing Parkinson's disease and may provide a way to distinguish variations within Parkinson's disease phenotype. The quantitative parameters however did not correlate with disease severity in Parkinson's disease. The assay distinguished multiple system atrophy patients from Parkinson's disease patients and in contrast to Parkinson's disease, the quantitative parameters correlated with disease progression of multiple system atrophy. Our results also provided further evidence for αSyn-RT-QuIC having potential as an early biomarker detecting synucleinopathy in idiopathic REM sleep behaviour disorder patients prior to conversion. Further analysis of longitudinally followed idiopathic REM sleep behaviour disorder patients is needed to better understand the relationship between αSyn-RT-QuIC signature and the progression from prodromal to different synucleinopathies.
dc.language.isoENen_US
dc.rightsAttribution-NonCommercial 3.0 United States*
dc.rights.urihttp://creativecommons.org/licenses/by-nc/3.0/us/*
dc.subject.enα-synuclein
dc.subject.enBiomarker
dc.subject.enProdromal
dc.subject.enSeeding
dc.subject.enStratification
dc.title.enDiagnostic value of cerebrospinal fluid alpha-synuclein seed quantification in synucleinopathies
dc.typeArticle de revueen_US
dc.identifier.doi10.1093/brain/awab431en_US
dc.subject.halSciences du Vivant [q-bio]/Santé publique et épidémiologieen_US
dc.identifier.pubmed34894214en_US
bordeaux.journalBrain - A Journal of Neurologyen_US
bordeaux.hal.laboratoriesBordeaux Population Health Research Center (BPH) - UMR 1219en_US
bordeaux.institutionUniversité de Bordeauxen_US
bordeaux.institutionINSERMen_US
bordeaux.teamSEPIAen_US
bordeaux.peerReviewedouien_US
bordeaux.inpressnonen_US
hal.identifierhal-03533847
hal.version1
hal.date.transferred2022-01-19T08:35:42Z
hal.exporttrue
dc.rights.ccPas de Licence CCen_US
bordeaux.COinSctx_ver=Z39.88-2004&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.jtitle=Brain%20-%20A%20Journal%20of%20Neurology&rft.date=2021-12-11&rft.eissn=1460-2156%20(Electronic)%200006-8950%20(Linking)&rft.issn=1460-2156%20(Electronic)%200006-8950%20(Linking)&rft.au=POGGIOLINI,%20Ilaria&GUPTA,%20Vandana&LAWTON,%20Michael&LEE,%20Seoyun&EL-TURABI,%20Aadil&rft.genre=article


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