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Gene, Stem Cell, and Alternative Therapies for SCA 1
Spinocerebellar ataxia 1 is an autosomal dominant disease characterized by neurodegeneration and motor dysfunction. In disease pathogenesis, polyglutamine expansion within Ataxin-1, a gene involved in transcriptional repression, causes protein nuclear inclusions to form. Most notably, neuronal dysfu...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2016
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4981596/ https://www.ncbi.nlm.nih.gov/pubmed/27570504 http://dx.doi.org/10.3389/fnmol.2016.00067 |
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author | Wagner, Jacob L. O'Connor, Deirdre M. Donsante, Anthony Boulis, Nicholas M. |
author_facet | Wagner, Jacob L. O'Connor, Deirdre M. Donsante, Anthony Boulis, Nicholas M. |
author_sort | Wagner, Jacob L. |
collection | PubMed |
description | Spinocerebellar ataxia 1 is an autosomal dominant disease characterized by neurodegeneration and motor dysfunction. In disease pathogenesis, polyglutamine expansion within Ataxin-1, a gene involved in transcriptional repression, causes protein nuclear inclusions to form. Most notably, neuronal dysfunction presents in Purkinje cells. However, the effect of mutant Ataxin-1 is not entirely understood. Two mouse models are employed to represent spinocerebellar ataxia 1, a B05 transgenic model that specifically expresses mutant Ataxin-1 in Purkinje cells, and a Sca1 154Q/2Q model that inserts the polyglutamine expansion into the mouse Ataxin-1 locus so that the mutant Ataxin-1 is expressed in all cells that express Ataxin-1. This review aims to summarize and evaluate the wide variety of therapies proposed for spinocerebellar ataxia 1, specifically gene and stem cell therapies. |
format | Online Article Text |
id | pubmed-4981596 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-49815962016-08-26 Gene, Stem Cell, and Alternative Therapies for SCA 1 Wagner, Jacob L. O'Connor, Deirdre M. Donsante, Anthony Boulis, Nicholas M. Front Mol Neurosci Neuroscience Spinocerebellar ataxia 1 is an autosomal dominant disease characterized by neurodegeneration and motor dysfunction. In disease pathogenesis, polyglutamine expansion within Ataxin-1, a gene involved in transcriptional repression, causes protein nuclear inclusions to form. Most notably, neuronal dysfunction presents in Purkinje cells. However, the effect of mutant Ataxin-1 is not entirely understood. Two mouse models are employed to represent spinocerebellar ataxia 1, a B05 transgenic model that specifically expresses mutant Ataxin-1 in Purkinje cells, and a Sca1 154Q/2Q model that inserts the polyglutamine expansion into the mouse Ataxin-1 locus so that the mutant Ataxin-1 is expressed in all cells that express Ataxin-1. This review aims to summarize and evaluate the wide variety of therapies proposed for spinocerebellar ataxia 1, specifically gene and stem cell therapies. Frontiers Media S.A. 2016-08-12 /pmc/articles/PMC4981596/ /pubmed/27570504 http://dx.doi.org/10.3389/fnmol.2016.00067 Text en Copyright © 2016 Wagner, O'Connor, Donsante and Boulis. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Neuroscience Wagner, Jacob L. O'Connor, Deirdre M. Donsante, Anthony Boulis, Nicholas M. Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title | Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title_full | Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title_fullStr | Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title_full_unstemmed | Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title_short | Gene, Stem Cell, and Alternative Therapies for SCA 1 |
title_sort | gene, stem cell, and alternative therapies for sca 1 |
topic | Neuroscience |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4981596/ https://www.ncbi.nlm.nih.gov/pubmed/27570504 http://dx.doi.org/10.3389/fnmol.2016.00067 |
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