Cargando…
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...
Autores principales: | , , , , , , , , , , , , , , , , , , , |
---|---|
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 |
_version_ | 1782436497931108352 |
---|---|
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. |
format | Online Article Text |
id | pubmed-4899632 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Nature Publishing Group |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT benraissabdellatif humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT wangsu humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT herrlingerstephanie humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT lixiaojie humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT chandlermilitellodevin humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT maucerijoseph humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT burmhayleyb humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT tonermichael humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT osipovitchmikhail humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT jimxuqiwu humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT dingfengfei humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT wangfushun humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT kangning humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT kangjian humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT curtinpaulc humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT brunnerdaniela humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT windremmarthas humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT munozsanjuanignacio humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT nedergaardmaiken humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease AT goldmanstevena humangliacanbothinduceandrescueaspectsofdiseasephenotypeinhuntingtondisease |