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Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice
Huntington's disease (HD) is an inherited neurodegenerative disorder with no disease-modifying treatment. Expansion of the glutamine-encoding repeat in the Huntingtin (HTT) gene causes broad effects that are a challenge for single treatment strategies. Strategies based on human stem cells offer...
Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Elsevier
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5768890/ https://www.ncbi.nlm.nih.gov/pubmed/29233555 http://dx.doi.org/10.1016/j.stemcr.2017.11.005 |
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author | Reidling, Jack C. Relaño-Ginés, Aroa Holley, Sandra M. Ochaba, Joseph Moore, Cindy Fury, Brian Lau, Alice Tran, Andrew H. Yeung, Sylvia Salamati, Delaram Zhu, Chunni Hatami, Asa Cepeda, Carlos Barry, Joshua A. Kamdjou, Talia King, Alvin Coleal-Bergum, Dane Franich, Nicholas R. LaFerla, Frank M. Steffan, Joan S. Blurton-Jones, Mathew Meshul, Charles K. Bauer, Gerhard Levine, Michael S. Chesselet, Marie-Francoise Thompson, Leslie M. |
author_facet | Reidling, Jack C. Relaño-Ginés, Aroa Holley, Sandra M. Ochaba, Joseph Moore, Cindy Fury, Brian Lau, Alice Tran, Andrew H. Yeung, Sylvia Salamati, Delaram Zhu, Chunni Hatami, Asa Cepeda, Carlos Barry, Joshua A. Kamdjou, Talia King, Alvin Coleal-Bergum, Dane Franich, Nicholas R. LaFerla, Frank M. Steffan, Joan S. Blurton-Jones, Mathew Meshul, Charles K. Bauer, Gerhard Levine, Michael S. Chesselet, Marie-Francoise Thompson, Leslie M. |
author_sort | Reidling, Jack C. |
collection | PubMed |
description | Huntington's disease (HD) is an inherited neurodegenerative disorder with no disease-modifying treatment. Expansion of the glutamine-encoding repeat in the Huntingtin (HTT) gene causes broad effects that are a challenge for single treatment strategies. Strategies based on human stem cells offer a promising option. We evaluated efficacy of transplanting a good manufacturing practice (GMP)-grade human embryonic stem cell-derived neural stem cell (hNSC) line into striatum of HD modeled mice. In HD fragment model R6/2 mice, transplants improve motor deficits, rescue synaptic alterations, and are contacted by nerve terminals from mouse cells. Furthermore, implanted hNSCs are electrophysiologically active. hNSCs also improved motor and late-stage cognitive impairment in a second HD model, Q140 knockin mice. Disease-modifying activity is suggested by the reduction of aberrant accumulation of mutant HTT protein and expression of brain-derived neurotrophic factor (BDNF) in both models. These findings hold promise for future development of stem cell-based therapies. |
format | Online Article Text |
id | pubmed-5768890 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | Elsevier |
record_format | MEDLINE/PubMed |
spelling | pubmed-57688902018-01-18 Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice Reidling, Jack C. Relaño-Ginés, Aroa Holley, Sandra M. Ochaba, Joseph Moore, Cindy Fury, Brian Lau, Alice Tran, Andrew H. Yeung, Sylvia Salamati, Delaram Zhu, Chunni Hatami, Asa Cepeda, Carlos Barry, Joshua A. Kamdjou, Talia King, Alvin Coleal-Bergum, Dane Franich, Nicholas R. LaFerla, Frank M. Steffan, Joan S. Blurton-Jones, Mathew Meshul, Charles K. Bauer, Gerhard Levine, Michael S. Chesselet, Marie-Francoise Thompson, Leslie M. Stem Cell Reports Article Huntington's disease (HD) is an inherited neurodegenerative disorder with no disease-modifying treatment. Expansion of the glutamine-encoding repeat in the Huntingtin (HTT) gene causes broad effects that are a challenge for single treatment strategies. Strategies based on human stem cells offer a promising option. We evaluated efficacy of transplanting a good manufacturing practice (GMP)-grade human embryonic stem cell-derived neural stem cell (hNSC) line into striatum of HD modeled mice. In HD fragment model R6/2 mice, transplants improve motor deficits, rescue synaptic alterations, and are contacted by nerve terminals from mouse cells. Furthermore, implanted hNSCs are electrophysiologically active. hNSCs also improved motor and late-stage cognitive impairment in a second HD model, Q140 knockin mice. Disease-modifying activity is suggested by the reduction of aberrant accumulation of mutant HTT protein and expression of brain-derived neurotrophic factor (BDNF) in both models. These findings hold promise for future development of stem cell-based therapies. Elsevier 2017-12-07 /pmc/articles/PMC5768890/ /pubmed/29233555 http://dx.doi.org/10.1016/j.stemcr.2017.11.005 Text en © 2017 The Authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). |
spellingShingle | Article Reidling, Jack C. Relaño-Ginés, Aroa Holley, Sandra M. Ochaba, Joseph Moore, Cindy Fury, Brian Lau, Alice Tran, Andrew H. Yeung, Sylvia Salamati, Delaram Zhu, Chunni Hatami, Asa Cepeda, Carlos Barry, Joshua A. Kamdjou, Talia King, Alvin Coleal-Bergum, Dane Franich, Nicholas R. LaFerla, Frank M. Steffan, Joan S. Blurton-Jones, Mathew Meshul, Charles K. Bauer, Gerhard Levine, Michael S. Chesselet, Marie-Francoise Thompson, Leslie M. Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title | Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title_full | Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title_fullStr | Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title_full_unstemmed | Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title_short | Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice |
title_sort | human neural stem cell transplantation rescues functional deficits in r6/2 and q140 huntington's disease mice |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5768890/ https://www.ncbi.nlm.nih.gov/pubmed/29233555 http://dx.doi.org/10.1016/j.stemcr.2017.11.005 |
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