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rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs

Krabbe disease is a rare neurodegenerative fatal disease. It is caused by deficiency of the lysosomal enzyme galactocerebrosidase (GALC), which results in progressive accumulation of galactolipid substrates in myelin-forming cells. However, there is still a lack of appropriate neural models and effe...

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Autores principales: Tian, Guoshuai, Cao, Chunyu, Li, Shuyue, Wang, Wei, Zhang, Ye, Lv, Yafeng
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10143348/
https://www.ncbi.nlm.nih.gov/pubmed/37111381
http://dx.doi.org/10.3390/ph16040624
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author Tian, Guoshuai
Cao, Chunyu
Li, Shuyue
Wang, Wei
Zhang, Ye
Lv, Yafeng
author_facet Tian, Guoshuai
Cao, Chunyu
Li, Shuyue
Wang, Wei
Zhang, Ye
Lv, Yafeng
author_sort Tian, Guoshuai
collection PubMed
description Krabbe disease is a rare neurodegenerative fatal disease. It is caused by deficiency of the lysosomal enzyme galactocerebrosidase (GALC), which results in progressive accumulation of galactolipid substrates in myelin-forming cells. However, there is still a lack of appropriate neural models and effective approaches for Krabbe disease. We generated induced pluripotent stem cells (iPSCs) from a Krabbe patient previously. Here, Krabbe patient-derived neural stem cells (K-NSCs) were induced from these iPSCs. By using nine kinds of recombinant adeno-associated virus (rAAV) vectors to infect K-NSCs, we found that the rAAV2 vector has high transduction efficiency for K-NSCs. Most importantly, rAAV2-GALC rescued GALC enzymatic activity in K-NSCs. Our findings not only establish a novel patient NSC model for Krabbe disease, but also firstly indicate the potential of rAAV2-mediated gene therapy for this devastating disease.
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spelling pubmed-101433482023-04-29 rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs Tian, Guoshuai Cao, Chunyu Li, Shuyue Wang, Wei Zhang, Ye Lv, Yafeng Pharmaceuticals (Basel) Article Krabbe disease is a rare neurodegenerative fatal disease. It is caused by deficiency of the lysosomal enzyme galactocerebrosidase (GALC), which results in progressive accumulation of galactolipid substrates in myelin-forming cells. However, there is still a lack of appropriate neural models and effective approaches for Krabbe disease. We generated induced pluripotent stem cells (iPSCs) from a Krabbe patient previously. Here, Krabbe patient-derived neural stem cells (K-NSCs) were induced from these iPSCs. By using nine kinds of recombinant adeno-associated virus (rAAV) vectors to infect K-NSCs, we found that the rAAV2 vector has high transduction efficiency for K-NSCs. Most importantly, rAAV2-GALC rescued GALC enzymatic activity in K-NSCs. Our findings not only establish a novel patient NSC model for Krabbe disease, but also firstly indicate the potential of rAAV2-mediated gene therapy for this devastating disease. MDPI 2023-04-20 /pmc/articles/PMC10143348/ /pubmed/37111381 http://dx.doi.org/10.3390/ph16040624 Text en © 2023 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 Article
Tian, Guoshuai
Cao, Chunyu
Li, Shuyue
Wang, Wei
Zhang, Ye
Lv, Yafeng
rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title_full rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title_fullStr rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title_full_unstemmed rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title_short rAAV2-Mediated Restoration of GALC in Neural Stem Cells from Krabbe Patient-Derived iPSCs
title_sort raav2-mediated restoration of galc in neural stem cells from krabbe patient-derived ipscs
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10143348/
https://www.ncbi.nlm.nih.gov/pubmed/37111381
http://dx.doi.org/10.3390/ph16040624
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