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Human glia can both induce and rescue aspects of disease phenotype in Huntington disease

The causal contribution of glial pathology to Huntington disease (HD) has not been heavily explored. To define the contribution of glia to HD, we established human HD glial chimeras by neonatally engrafting immunodeficient mice with mutant huntingtin (mHTT)-expressing human glial progenitor cells (h...

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Autores principales: Benraiss, Abdellatif, Wang, Su, Herrlinger, Stephanie, Li, Xiaojie, Chandler-Militello, Devin, Mauceri, Joseph, Burm, Hayley B., Toner, Michael, Osipovitch, Mikhail, Jim Xu, Qiwu, Ding, Fengfei, Wang, Fushun, Kang, Ning, Kang, Jian, Curtin, Paul C., Brunner, Daniela, Windrem, Martha S., Munoz-Sanjuan, Ignacio, Nedergaard, Maiken, Goldman, Steven A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4899632/
https://www.ncbi.nlm.nih.gov/pubmed/27273432
http://dx.doi.org/10.1038/ncomms11758
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author Benraiss, Abdellatif
Wang, Su
Herrlinger, Stephanie
Li, Xiaojie
Chandler-Militello, Devin
Mauceri, Joseph
Burm, Hayley B.
Toner, Michael
Osipovitch, Mikhail
Jim Xu, Qiwu
Ding, Fengfei
Wang, Fushun
Kang, Ning
Kang, Jian
Curtin, Paul C.
Brunner, Daniela
Windrem, Martha S.
Munoz-Sanjuan, Ignacio
Nedergaard, Maiken
Goldman, Steven A.
author_facet Benraiss, Abdellatif
Wang, Su
Herrlinger, Stephanie
Li, Xiaojie
Chandler-Militello, Devin
Mauceri, Joseph
Burm, Hayley B.
Toner, Michael
Osipovitch, Mikhail
Jim Xu, Qiwu
Ding, Fengfei
Wang, Fushun
Kang, Ning
Kang, Jian
Curtin, Paul C.
Brunner, Daniela
Windrem, Martha S.
Munoz-Sanjuan, Ignacio
Nedergaard, Maiken
Goldman, Steven A.
author_sort Benraiss, Abdellatif
collection PubMed
description The causal contribution of glial pathology to Huntington disease (HD) has not been heavily explored. To define the contribution of glia to HD, we established human HD glial chimeras by neonatally engrafting immunodeficient mice with mutant huntingtin (mHTT)-expressing human glial progenitor cells (hGPCs), derived from either human embryonic stem cells or mHTT-transduced fetal hGPCs. Here we show that mHTT glia can impart disease phenotype to normal mice, since mice engrafted intrastriatally with mHTT hGPCs exhibit worse motor performance than controls, and striatal neurons in mHTT glial chimeras are hyperexcitable. Conversely, normal glia can ameliorate disease phenotype in transgenic HD mice, as striatal transplantation of normal glia rescues aspects of electrophysiological and behavioural phenotype, restores interstitial potassium homeostasis, slows disease progression and extends survival in R6/2 HD mice. These observations suggest a causal role for glia in HD, and further suggest a cell-based strategy for disease amelioration in this disorder.
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spelling pubmed-48996322016-06-22 Human glia can both induce and rescue aspects of disease phenotype in Huntington disease Benraiss, Abdellatif Wang, Su Herrlinger, Stephanie Li, Xiaojie Chandler-Militello, Devin Mauceri, Joseph Burm, Hayley B. Toner, Michael Osipovitch, Mikhail Jim Xu, Qiwu Ding, Fengfei Wang, Fushun Kang, Ning Kang, Jian Curtin, Paul C. Brunner, Daniela Windrem, Martha S. Munoz-Sanjuan, Ignacio Nedergaard, Maiken Goldman, Steven A. Nat Commun Article The causal contribution of glial pathology to Huntington disease (HD) has not been heavily explored. To define the contribution of glia to HD, we established human HD glial chimeras by neonatally engrafting immunodeficient mice with mutant huntingtin (mHTT)-expressing human glial progenitor cells (hGPCs), derived from either human embryonic stem cells or mHTT-transduced fetal hGPCs. Here we show that mHTT glia can impart disease phenotype to normal mice, since mice engrafted intrastriatally with mHTT hGPCs exhibit worse motor performance than controls, and striatal neurons in mHTT glial chimeras are hyperexcitable. Conversely, normal glia can ameliorate disease phenotype in transgenic HD mice, as striatal transplantation of normal glia rescues aspects of electrophysiological and behavioural phenotype, restores interstitial potassium homeostasis, slows disease progression and extends survival in R6/2 HD mice. These observations suggest a causal role for glia in HD, and further suggest a cell-based strategy for disease amelioration in this disorder. Nature Publishing Group 2016-06-07 /pmc/articles/PMC4899632/ /pubmed/27273432 http://dx.doi.org/10.1038/ncomms11758 Text en Copyright © 2016, Nature Publishing Group, a division of Macmillan Publishers Limited. All Rights Reserved. http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article's Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/
spellingShingle Article
Benraiss, Abdellatif
Wang, Su
Herrlinger, Stephanie
Li, Xiaojie
Chandler-Militello, Devin
Mauceri, Joseph
Burm, Hayley B.
Toner, Michael
Osipovitch, Mikhail
Jim Xu, Qiwu
Ding, Fengfei
Wang, Fushun
Kang, Ning
Kang, Jian
Curtin, Paul C.
Brunner, Daniela
Windrem, Martha S.
Munoz-Sanjuan, Ignacio
Nedergaard, Maiken
Goldman, Steven A.
Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title_full Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title_fullStr Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title_full_unstemmed Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title_short Human glia can both induce and rescue aspects of disease phenotype in Huntington disease
title_sort human glia can both induce and rescue aspects of disease phenotype in huntington disease
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4899632/
https://www.ncbi.nlm.nih.gov/pubmed/27273432
http://dx.doi.org/10.1038/ncomms11758
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