Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?

Introduction: the MDS-PSP criteria have shown high sensitivity for the PSP diagnosis, but do not discriminate the phenotype diversity. Our purpose was to search for anatomopathological differences among PSP phenotypes resulting from the application of the MDS-PSP criteria comparing with the previous...

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Autores: Sánchez Ruiz de Gordoa, Javier, Zelaya Huerta, María Victoria, Tellechea-Aramburo, Paula, Acha Santamaría, Blanca, Roldán, Miren, López Molina, Carlos, Coca, Valle, Galbete Jiménez, Arkaitz, Mendióroz Iriarte, Maite, Erro Aguirre, María Elena
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2022
País:España
Institución:Universidad Pública de Navarra
Repositorio:Academica-e. Repositorio Institucional de la Universidad Pública de Navarra
OAI Identifier:oai:academica-e.unavarra.es:2454/43365
Acceso en línea:https://hdl.handle.net/2454/43365
Access Level:acceso abierto
Palabra clave:Clinical-pathological correlation
Phenotypes
Progressive supranuclear palsy (PSP)
PSP-MDS criteria
Tau protein load
Tauopathies
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spelling Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?Sánchez Ruiz de Gordoa, JavierZelaya Huerta, María VictoriaTellechea-Aramburo, PaulaAcha Santamaría, BlancaRoldán, MirenLópez Molina, CarlosCoca, ValleGalbete Jiménez, ArkaitzMendióroz Iriarte, MaiteErro Aguirre, María ElenaClinical-pathological correlationPhenotypesProgressive supranuclear palsy (PSP)PSP-MDS criteriaTau protein loadTauopathiesIntroduction: the MDS-PSP criteria have shown high sensitivity for the PSP diagnosis, but do not discriminate the phenotype diversity. Our purpose was to search for anatomopathological differences among PSP phenotypes resulting from the application of the MDS-PSP criteria comparing with the previous ones. Methods: thirty-four PSP cases from a single brain bank were retrospectively classified according to the criteria used by Respondek et al. in 2014 and the PSP-MDS criteria at 3 years (MDS-3y), 6 years (MDS-6y) and at the last clinical evaluation before death (MDS-last). Semiquantitative measurement of total, cortical and subcortical tau load was compared. For comparative analysis, PSP-Richardson syndrome and PSP postural instability were grouped (PSP-RS/PI) as well as the PSP atypical cortical phenotypes (PSP-Cx). Results: applying the Respondek's criteria, PSP phenotypes were distributed as follow: 55.9% PSP-RS/PI, 26.5% PSP-Cx, 11.8% PSP-Parkinsonism (PSP-P), and 5.9% PSP-Cerebellum. PSP-RS/PI and PSP-Cx had a higher total tau load than PSP-P; PSP-Cx showed a higher cortical tau load than PSP-RS/PI and PSP-P; and PSP-RS/PI had a higher subcortical tau load than PSP-P. Applying the MDS-3y, MDS-6y and MDS-last criteria; the PSP-RS/PI group increased (67.6, 70.6 and 70.6% respectively) whereas the PSP-Cx group decreased (8.8, and 8.8 and 11.8%). Then, only differences in total and subcortical tau burden between PSP-RS/PI and PSP-P were observed. Interpretation: after the retrospective application of the new MDS-PSP criteria, total and subcortical tau load is higher in PSP-RS/PI than in PSP-P whereas no other differences in tau load between phenotypes were found, as a consequence of the loss of phenotypic diversity.Frontiers MediaEstadística, Informática y MatemáticasEstatistika, Informatika eta Matematika2022info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionapplication/pdfhttps://hdl.handle.net/2454/43365reponame:Academica-e. Repositorio Institucional de la Universidad Pública de Navarrainstname:Universidad Pública de NavarraIngléshttps://creativecommons.org/licenses/by/4.0/info:eu-repo/semantics/openAccessoai:academica-e.unavarra.es:2454/433652026-06-17T12:41:47Z
dc.title.none.fl_str_mv Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
title Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
spellingShingle Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
Sánchez Ruiz de Gordoa, Javier
Clinical-pathological correlation
Phenotypes
Progressive supranuclear palsy (PSP)
PSP-MDS criteria
Tau protein load
Tauopathies
title_short Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
title_full Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
title_fullStr Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
title_full_unstemmed Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
title_sort Is the phenotype designation by PSP-MDS criteria stable throughout the disease course and consistent with tau distribution?
dc.creator.none.fl_str_mv Sánchez Ruiz de Gordoa, Javier
Zelaya Huerta, María Victoria
Tellechea-Aramburo, Paula
Acha Santamaría, Blanca
Roldán, Miren
López Molina, Carlos
Coca, Valle
Galbete Jiménez, Arkaitz
Mendióroz Iriarte, Maite
Erro Aguirre, María Elena
author Sánchez Ruiz de Gordoa, Javier
author_facet Sánchez Ruiz de Gordoa, Javier
Zelaya Huerta, María Victoria
Tellechea-Aramburo, Paula
Acha Santamaría, Blanca
Roldán, Miren
López Molina, Carlos
Coca, Valle
Galbete Jiménez, Arkaitz
Mendióroz Iriarte, Maite
Erro Aguirre, María Elena
author_role author
author2 Zelaya Huerta, María Victoria
Tellechea-Aramburo, Paula
Acha Santamaría, Blanca
Roldán, Miren
López Molina, Carlos
Coca, Valle
Galbete Jiménez, Arkaitz
Mendióroz Iriarte, Maite
Erro Aguirre, María Elena
author2_role author
author
author
author
author
author
author
author
author
dc.contributor.none.fl_str_mv Estadística, Informática y Matemáticas
Estatistika, Informatika eta Matematika
dc.subject.none.fl_str_mv Clinical-pathological correlation
Phenotypes
Progressive supranuclear palsy (PSP)
PSP-MDS criteria
Tau protein load
Tauopathies
topic Clinical-pathological correlation
Phenotypes
Progressive supranuclear palsy (PSP)
PSP-MDS criteria
Tau protein load
Tauopathies
description Introduction: the MDS-PSP criteria have shown high sensitivity for the PSP diagnosis, but do not discriminate the phenotype diversity. Our purpose was to search for anatomopathological differences among PSP phenotypes resulting from the application of the MDS-PSP criteria comparing with the previous ones. Methods: thirty-four PSP cases from a single brain bank were retrospectively classified according to the criteria used by Respondek et al. in 2014 and the PSP-MDS criteria at 3 years (MDS-3y), 6 years (MDS-6y) and at the last clinical evaluation before death (MDS-last). Semiquantitative measurement of total, cortical and subcortical tau load was compared. For comparative analysis, PSP-Richardson syndrome and PSP postural instability were grouped (PSP-RS/PI) as well as the PSP atypical cortical phenotypes (PSP-Cx). Results: applying the Respondek's criteria, PSP phenotypes were distributed as follow: 55.9% PSP-RS/PI, 26.5% PSP-Cx, 11.8% PSP-Parkinsonism (PSP-P), and 5.9% PSP-Cerebellum. PSP-RS/PI and PSP-Cx had a higher total tau load than PSP-P; PSP-Cx showed a higher cortical tau load than PSP-RS/PI and PSP-P; and PSP-RS/PI had a higher subcortical tau load than PSP-P. Applying the MDS-3y, MDS-6y and MDS-last criteria; the PSP-RS/PI group increased (67.6, 70.6 and 70.6% respectively) whereas the PSP-Cx group decreased (8.8, and 8.8 and 11.8%). Then, only differences in total and subcortical tau burden between PSP-RS/PI and PSP-P were observed. Interpretation: after the retrospective application of the new MDS-PSP criteria, total and subcortical tau load is higher in PSP-RS/PI than in PSP-P whereas no other differences in tau load between phenotypes were found, as a consequence of the loss of phenotypic diversity.
publishDate 2022
dc.date.none.fl_str_mv 2022
dc.type.none.fl_str_mv info:eu-repo/semantics/article
info:eu-repo/semantics/publishedVersion
format article
status_str publishedVersion
dc.identifier.none.fl_str_mv https://hdl.handle.net/2454/43365
url https://hdl.handle.net/2454/43365
dc.language.none.fl_str_mv Inglés
language_invalid_str_mv Inglés
dc.rights.none.fl_str_mv https://creativecommons.org/licenses/by/4.0/
info:eu-repo/semantics/openAccess
rights_invalid_str_mv https://creativecommons.org/licenses/by/4.0/
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.publisher.none.fl_str_mv Frontiers Media
publisher.none.fl_str_mv Frontiers Media
dc.source.none.fl_str_mv reponame:Academica-e. Repositorio Institucional de la Universidad Pública de Navarra
instname:Universidad Pública de Navarra
instname_str Universidad Pública de Navarra
reponame_str Academica-e. Repositorio Institucional de la Universidad Pública de Navarra
collection Academica-e. Repositorio Institucional de la Universidad Pública de Navarra
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