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Genetic Approaches for the Treatment of Giant Axonal Neuropathy
Giant axonal neuropathy (GAN) is a pediatric, hereditary, neurodegenerative disorder that affects both the central and peripheral nervous systems. It is caused by mutations in the GAN gene, which codes for the gigaxonin protein. Gigaxonin plays a role in intermediate filament (IF) turnover hence los...
Autores principales: | , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9865904/ https://www.ncbi.nlm.nih.gov/pubmed/36675752 http://dx.doi.org/10.3390/jpm13010091 |
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author | Shirakaki, Satomi Roshmi, Rohini Roy Yokota, Toshifumi |
author_facet | Shirakaki, Satomi Roshmi, Rohini Roy Yokota, Toshifumi |
author_sort | Shirakaki, Satomi |
collection | PubMed |
description | Giant axonal neuropathy (GAN) is a pediatric, hereditary, neurodegenerative disorder that affects both the central and peripheral nervous systems. It is caused by mutations in the GAN gene, which codes for the gigaxonin protein. Gigaxonin plays a role in intermediate filament (IF) turnover hence loss of function of this protein leads to IF aggregates in various types of cells. These aggregates can lead to abnormal cellular function that manifests as a diverse set of symptoms in persons with GAN including nerve degeneration, cognitive issues, skin diseases, vision loss, and muscle weakness. GAN has no cure at this time. Currently, an adeno-associated virus (AAV) 9-mediated gene replacement therapy is being tested in a phase I clinical trial for the treatment of GAN. This review paper aims to provide an overview of giant axonal neuropathy and the current efforts at developing a treatment for this devastating disease. |
format | Online Article Text |
id | pubmed-9865904 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-98659042023-01-22 Genetic Approaches for the Treatment of Giant Axonal Neuropathy Shirakaki, Satomi Roshmi, Rohini Roy Yokota, Toshifumi J Pers Med Review Giant axonal neuropathy (GAN) is a pediatric, hereditary, neurodegenerative disorder that affects both the central and peripheral nervous systems. It is caused by mutations in the GAN gene, which codes for the gigaxonin protein. Gigaxonin plays a role in intermediate filament (IF) turnover hence loss of function of this protein leads to IF aggregates in various types of cells. These aggregates can lead to abnormal cellular function that manifests as a diverse set of symptoms in persons with GAN including nerve degeneration, cognitive issues, skin diseases, vision loss, and muscle weakness. GAN has no cure at this time. Currently, an adeno-associated virus (AAV) 9-mediated gene replacement therapy is being tested in a phase I clinical trial for the treatment of GAN. This review paper aims to provide an overview of giant axonal neuropathy and the current efforts at developing a treatment for this devastating disease. MDPI 2022-12-30 /pmc/articles/PMC9865904/ /pubmed/36675752 http://dx.doi.org/10.3390/jpm13010091 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Review Shirakaki, Satomi Roshmi, Rohini Roy Yokota, Toshifumi Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title | Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title_full | Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title_fullStr | Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title_full_unstemmed | Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title_short | Genetic Approaches for the Treatment of Giant Axonal Neuropathy |
title_sort | genetic approaches for the treatment of giant axonal neuropathy |
topic | Review |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9865904/ https://www.ncbi.nlm.nih.gov/pubmed/36675752 http://dx.doi.org/10.3390/jpm13010091 |
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