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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...

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Autores principales: 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.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2017
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.
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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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