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Spreading in ALS: The relative impact of upper and lower motor neuron involvement

2020Artigo científico Acesso aberto
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dc.contributor.authorGromicho, Marta
dc.contributor.authorFigueiral, Manuel
dc.contributor.authorUysal, Hilmi
dc.contributor.authorGrosskreutz, Julian
dc.contributor.authorKuzma‐Kozakiewicz, Magdalena
dc.contributor.authorPinto, Susana
dc.contributor.authorPetri, Susanne
dc.contributor.authorMadeira, Sara C.
dc.contributor.authorSwash, Michael
dc.contributor.authorCarvalho, Mamede
dc.date.accessioned2022-06-06T15:01:17Z
dc.date.available2022-06-06T15:01:17Z
dc.date.issued2020
dc.description© 2020 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association. This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made.pt_PT
dc.description.abstractObjective: To investigate disease spread in amyotrophic lateral sclerosis (ALS), and determine the influence of lower (LMN) and upper motor neuron (UMN) involvement. Methods: We assessed disease spread in ALS in 1376 consecutively studied patients, from five European centers, applying an agreed proforma to assess LMN and UMN signs. We defined the pattern of disease onset and progression from predominant UMN or lower motor neuron (LMN) dysfunction in bulbar, upper limbs, lower limbs, and thoracic regions Non-linear regression analysis was applied to fit the data to a model that described the relation between two random variables, graphically represented by an inverse exponential curve. We analyzed the probability, rate of spread, and both combined (area under the curve). Results: We found that progression was more likely and quicker to or from the region of onset to close spinal regions. When the disease had a limb onset, bulbar motor neurons were more resistant. Furthermore, in the same time frame more patients progressed from bulbar to lower limbs than vice-versa, whether predominantly UMN or LMN involvement. Patients with initial thoracic involvement had a higher probability for rapid change. The presence of predominant UMN signs was associated with a faster caudal progression. Interpretation: Contiguous progression was leading pattern, and predominant UMN involvement is important in shortening the time for cranial-caudal spread. Our results can best be fitted to a model of independent LMN and UMN degeneration, with regional progression of LMN degeneration mostly by contiguity. UMN lesion causes an acceleration of rostral-caudal LMN loss.pt_PT
dc.description.sponsorshipThis is an EU Joint Programme - Neurodegenerative Disease Research (JPND) project. The project is supported through national funding organizations under the aegis of JPND - www.jpnd.eu. This project was also partially supported by FCT funding to Neuroclinomics2 (PTDC/EEI-SII/1937/2014).pt_PT
dc.description.versioninfo:eu-repo/semantics/publishedVersionpt_PT
dc.identifier.citationAnn Clin Transl Neurol. 2020 Jul;7(7):1181-1192pt_PT
dc.identifier.doi10.1002/acn3.51098pt_PT
dc.identifier.eissn2328-9503
dc.identifier.urihttp://hdl.handle.net/10451/53289
dc.language.isoengpt_PT
dc.peerreviewedyespt_PT
dc.publisherWileypt_PT
dc.relationNEUROCLINOMICS2 - Unravelling Prognostic Markers in NEUROdegenerative diseases through CLINical and OMICS data integration
dc.relation.publisherversionhttps://onlinelibrary.wiley.com/journal/23289503pt_PT
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/pt_PT
dc.titleSpreading in ALS: The relative impact of upper and lower motor neuron involvementpt_PT
dc.typejournal article
dspace.entity.typePublication
oaire.awardNumberPTDC/EEI-SII/1937/2014
oaire.awardTitleNEUROCLINOMICS2 - Unravelling Prognostic Markers in NEUROdegenerative diseases through CLINical and OMICS data integration
oaire.awardURIinfo:eu-repo/grantAgreement/FCT/3599-PPCDT/PTDC%2FEEI-SII%2F1937%2F2014/PT
oaire.citation.endPage1192pt_PT
oaire.citation.issue7pt_PT
oaire.citation.startPage1181pt_PT
oaire.citation.titleAnnals of Clinical and Translational Neurologypt_PT
oaire.citation.volume7pt_PT
oaire.fundingStream3599-PPCDT
person.familyNameSilva
person.familyNamePinto
person.familyNameMadeira
person.familyNameSwash
person.familyNamede Carvalho
person.givenNameMarta Luísa Gromicho Morgado
person.givenNameSusana
person.givenNameSara
person.givenNameMichael
person.givenNameMamede
person.identifierB-2171-2018
person.identifier593760
person.identifier.ciencia-idEA1D-1979-5C3A
person.identifier.ciencia-idD31B-E89C-CB94
person.identifier.ciencia-idAF12-AA0D-0C7B
person.identifier.orcid0000-0003-2111-4579
person.identifier.orcid0000-0002-0727-5897
person.identifier.orcid0000-0002-1459-8096
person.identifier.orcid0000-0002-8717-8914
person.identifier.orcid0000-0001-7556-0158
person.identifier.ridL-8394-2019
person.identifier.ridC-5494-2008
person.identifier.scopus-author-id6506583274
person.identifier.scopus-author-id23397978500
person.identifier.scopus-author-id6602138051
person.identifier.scopus-author-id7101990692
person.identifier.scopus-author-id7101893769
project.funder.identifierhttp://doi.org/10.13039/501100001871
project.funder.nameFundação para a Ciência e a Tecnologia
rcaap.rightsopenAccesspt_PT
rcaap.typearticlept_PT
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