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WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells

Mutations in WD40-repeat protein 62 (WDR62) are commonly associated with primary microcephaly and other developmental cortical malformations. We used human pluripotent stem cells (hPSC) to examine WDR62 function during human neural differentiation and model early stages of human corticogenesis. Neur...

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Autores principales: Alshawaf, Abdullah J., Antonic, Ana, Skafidas, Efstratios, Ng, Dominic Chi-Hung, Dottori, Mirella
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5485354/
https://www.ncbi.nlm.nih.gov/pubmed/28690640
http://dx.doi.org/10.1155/2017/7848932
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author Alshawaf, Abdullah J.
Antonic, Ana
Skafidas, Efstratios
Ng, Dominic Chi-Hung
Dottori, Mirella
author_facet Alshawaf, Abdullah J.
Antonic, Ana
Skafidas, Efstratios
Ng, Dominic Chi-Hung
Dottori, Mirella
author_sort Alshawaf, Abdullah J.
collection PubMed
description Mutations in WD40-repeat protein 62 (WDR62) are commonly associated with primary microcephaly and other developmental cortical malformations. We used human pluripotent stem cells (hPSC) to examine WDR62 function during human neural differentiation and model early stages of human corticogenesis. Neurospheres lacking WDR62 expression showed decreased expression of intermediate progenitor marker, TBR2, and also glial marker, S100β. In contrast, inhibition of c-Jun N-terminal kinase (JNK) signalling during hPSC neural differentiation induced upregulation of WDR62 with a corresponding increase in neural and glial progenitor markers, PAX6 and EAAT1, respectively. These findings may signify a role of WDR62 in specifying intermediate neural and glial progenitors during human pluripotent stem cell differentiation.
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spelling pubmed-54853542017-07-09 WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells Alshawaf, Abdullah J. Antonic, Ana Skafidas, Efstratios Ng, Dominic Chi-Hung Dottori, Mirella Stem Cells Int Research Article Mutations in WD40-repeat protein 62 (WDR62) are commonly associated with primary microcephaly and other developmental cortical malformations. We used human pluripotent stem cells (hPSC) to examine WDR62 function during human neural differentiation and model early stages of human corticogenesis. Neurospheres lacking WDR62 expression showed decreased expression of intermediate progenitor marker, TBR2, and also glial marker, S100β. In contrast, inhibition of c-Jun N-terminal kinase (JNK) signalling during hPSC neural differentiation induced upregulation of WDR62 with a corresponding increase in neural and glial progenitor markers, PAX6 and EAAT1, respectively. These findings may signify a role of WDR62 in specifying intermediate neural and glial progenitors during human pluripotent stem cell differentiation. Hindawi 2017 2017-06-13 /pmc/articles/PMC5485354/ /pubmed/28690640 http://dx.doi.org/10.1155/2017/7848932 Text en Copyright © 2017 Abdullah J. Alshawaf et al. https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Alshawaf, Abdullah J.
Antonic, Ana
Skafidas, Efstratios
Ng, Dominic Chi-Hung
Dottori, Mirella
WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title_full WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title_fullStr WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title_full_unstemmed WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title_short WDR62 Regulates Early Neural and Glial Progenitor Specification of Human Pluripotent Stem Cells
title_sort wdr62 regulates early neural and glial progenitor specification of human pluripotent stem cells
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5485354/
https://www.ncbi.nlm.nih.gov/pubmed/28690640
http://dx.doi.org/10.1155/2017/7848932
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