Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Wagner, Jacob L., O'Connor, Deirdre M., Donsante, Anthony, Boulis, Nicholas M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2016
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
_version_ 1782447633172791296
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
work_keys_str_mv AT wagnerjacobl genestemcellandalternativetherapiesforsca1
AT oconnordeirdrem genestemcellandalternativetherapiesforsca1
AT donsanteanthony genestemcellandalternativetherapiesforsca1
AT boulisnicholasm genestemcellandalternativetherapiesforsca1