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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...

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Detalles Bibliográficos
Autores principales: Sivakumar, Anusha, Cherqui, Stephanie
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
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.
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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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