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In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons

Impairment of GABAergic inhibitory neuronal function is linked to epilepsy and other neurological and psychiatric disorders. Recombinant adeno-associated virus (rAAV)-based gene therapy targeting GABAergic neurons is a promising treatment for GABA-associated disorders. However, there is a need to de...

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Autores principales: Niibori, Yosuke, Duba-Kiss, Robert, Bruder, Joseph T., Smith, Jared B., Hampson, David R.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Society of Gene & Cell Therapy 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9974971/
https://www.ncbi.nlm.nih.gov/pubmed/36874244
http://dx.doi.org/10.1016/j.omtm.2023.01.007
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author Niibori, Yosuke
Duba-Kiss, Robert
Bruder, Joseph T.
Smith, Jared B.
Hampson, David R.
author_facet Niibori, Yosuke
Duba-Kiss, Robert
Bruder, Joseph T.
Smith, Jared B.
Hampson, David R.
author_sort Niibori, Yosuke
collection PubMed
description Impairment of GABAergic inhibitory neuronal function is linked to epilepsy and other neurological and psychiatric disorders. Recombinant adeno-associated virus (rAAV)-based gene therapy targeting GABAergic neurons is a promising treatment for GABA-associated disorders. However, there is a need to develop rAAV-compatible gene-regulatory elements capable of selectively driving expression in GABAergic neurons throughout the brain. Here, we designed several novel GABAergic gene promoters. In silico analyses, including evolutionarily conserved DNA sequence alignments and transcription factor binding site searches among GABAergic neuronal genes, were carried out to reveal novel sequences for use as rAAV-compatible promoters. rAAVs (serotype 9) were injected into the CSF of neonatal mice and into the brain parenchyma of adult mice to assess promoter specificity. In mice injected neonatally, transgene expression was detected in multiple brain regions with very high neuronal specificity and moderate-to-high GABAergic neuronal selectivity. The GABA promoters differed greatly in their levels of expression and, in some brain regions, showed strikingly different patterns of GABAergic neuron transduction. This study is the first report of rAAV vectors that are functional in multiple brain regions using promoters designed by in silico analyses from multiple GABAergic genes. These novel GABA-targeting vectors may be useful tools to advance gene therapy for GABA-associated disorders.
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spelling pubmed-99749712023-03-02 In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons Niibori, Yosuke Duba-Kiss, Robert Bruder, Joseph T. Smith, Jared B. Hampson, David R. Mol Ther Methods Clin Dev Original Article Impairment of GABAergic inhibitory neuronal function is linked to epilepsy and other neurological and psychiatric disorders. Recombinant adeno-associated virus (rAAV)-based gene therapy targeting GABAergic neurons is a promising treatment for GABA-associated disorders. However, there is a need to develop rAAV-compatible gene-regulatory elements capable of selectively driving expression in GABAergic neurons throughout the brain. Here, we designed several novel GABAergic gene promoters. In silico analyses, including evolutionarily conserved DNA sequence alignments and transcription factor binding site searches among GABAergic neuronal genes, were carried out to reveal novel sequences for use as rAAV-compatible promoters. rAAVs (serotype 9) were injected into the CSF of neonatal mice and into the brain parenchyma of adult mice to assess promoter specificity. In mice injected neonatally, transgene expression was detected in multiple brain regions with very high neuronal specificity and moderate-to-high GABAergic neuronal selectivity. The GABA promoters differed greatly in their levels of expression and, in some brain regions, showed strikingly different patterns of GABAergic neuron transduction. This study is the first report of rAAV vectors that are functional in multiple brain regions using promoters designed by in silico analyses from multiple GABAergic genes. These novel GABA-targeting vectors may be useful tools to advance gene therapy for GABA-associated disorders. American Society of Gene & Cell Therapy 2023-02-04 /pmc/articles/PMC9974971/ /pubmed/36874244 http://dx.doi.org/10.1016/j.omtm.2023.01.007 Text en © 2023 The Authors https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Original Article
Niibori, Yosuke
Duba-Kiss, Robert
Bruder, Joseph T.
Smith, Jared B.
Hampson, David R.
In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title_full In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title_fullStr In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title_full_unstemmed In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title_short In silico prediction and in vivo testing of promoters targeting GABAergic inhibitory neurons
title_sort in silico prediction and in vivo testing of promoters targeting gabaergic inhibitory neurons
topic Original Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9974971/
https://www.ncbi.nlm.nih.gov/pubmed/36874244
http://dx.doi.org/10.1016/j.omtm.2023.01.007
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