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Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia
Friedreich’s ataxia (FRDA) is an inherited, multisystemic disorder predominantly caused by GAA hyper expansion in intron 1 of frataxin (FXN) gene. This expansion mutation transcriptionally represses FXN, a mitochondrial protein that is required for iron metabolism and mitochondrial homeostasis, lead...
Autores principales: | , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2022
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9157421/ https://www.ncbi.nlm.nih.gov/pubmed/35663795 http://dx.doi.org/10.3389/fgeed.2022.903139 |
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author | Sivakumar, Anusha Cherqui, Stephanie |
author_facet | Sivakumar, Anusha Cherqui, Stephanie |
author_sort | Sivakumar, Anusha |
collection | PubMed |
description | Friedreich’s ataxia (FRDA) is an inherited, multisystemic disorder predominantly caused by GAA hyper expansion in intron 1 of frataxin (FXN) gene. This expansion mutation transcriptionally represses FXN, a mitochondrial protein that is required for iron metabolism and mitochondrial homeostasis, leading to neurodegerative and cardiac dysfunction. Current therapeutic options for FRDA are focused on improving mitochondrial function and increasing frataxin expression through pharmacological interventions but are not effective in delaying or preventing the neurodegeneration in clinical trials. Recent research on in vivo and ex vivo gene therapy methods in FRDA animal and cell models showcase its promise as a one-time therapy for FRDA. In this review, we provide an overview on the current and emerging prospects of gene therapy for FRDA, with specific focus on advantages of CRISPR/Cas9-mediated gene editing of FXN as a viable option to restore endogenous frataxin expression. We also assess the potential of ex vivo gene editing in hematopoietic stem and progenitor cells as a potential autologous transplantation therapeutic option and discuss its advantages in tackling FRDA-specific safety aspects for clinical translation. |
format | Online Article Text |
id | pubmed-9157421 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-91574212022-06-02 Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia Sivakumar, Anusha Cherqui, Stephanie Front Genome Ed Genome Editing Friedreich’s ataxia (FRDA) is an inherited, multisystemic disorder predominantly caused by GAA hyper expansion in intron 1 of frataxin (FXN) gene. This expansion mutation transcriptionally represses FXN, a mitochondrial protein that is required for iron metabolism and mitochondrial homeostasis, leading to neurodegerative and cardiac dysfunction. Current therapeutic options for FRDA are focused on improving mitochondrial function and increasing frataxin expression through pharmacological interventions but are not effective in delaying or preventing the neurodegeneration in clinical trials. Recent research on in vivo and ex vivo gene therapy methods in FRDA animal and cell models showcase its promise as a one-time therapy for FRDA. In this review, we provide an overview on the current and emerging prospects of gene therapy for FRDA, with specific focus on advantages of CRISPR/Cas9-mediated gene editing of FXN as a viable option to restore endogenous frataxin expression. We also assess the potential of ex vivo gene editing in hematopoietic stem and progenitor cells as a potential autologous transplantation therapeutic option and discuss its advantages in tackling FRDA-specific safety aspects for clinical translation. Frontiers Media S.A. 2022-05-17 /pmc/articles/PMC9157421/ /pubmed/35663795 http://dx.doi.org/10.3389/fgeed.2022.903139 Text en Copyright © 2022 Sivakumar and Cherqui. 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) and the copyright owner(s) 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 | Genome Editing Sivakumar, Anusha Cherqui, Stephanie Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title | Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title_full | Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title_fullStr | Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title_full_unstemmed | Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title_short | Advantages and Limitations of Gene Therapy and Gene Editing for Friedreich’s Ataxia |
title_sort | advantages and limitations of gene therapy and gene editing for friedreich’s ataxia |
topic | Genome Editing |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9157421/ https://www.ncbi.nlm.nih.gov/pubmed/35663795 http://dx.doi.org/10.3389/fgeed.2022.903139 |
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