Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Shirakaki, Satomi, Roshmi, Rohini Roy, Yokota, Toshifumi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
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
_version_ 1784875955497467904
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
work_keys_str_mv AT shirakakisatomi geneticapproachesforthetreatmentofgiantaxonalneuropathy
AT roshmirohiniroy geneticapproachesforthetreatmentofgiantaxonalneuropathy
AT yokotatoshifumi geneticapproachesforthetreatmentofgiantaxonalneuropathy