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Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea

Connexins are gap junction components that are essential for acquiring normal hearing ability. Up to 50% of congenital, autosomal-recessive, non-syndromic deafness can be attributed to variants in GJB2, the gene that encodes connexin 26. Gene therapies modifying the expression of connexins are a fea...

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Autores principales: Hosoya, Makoto, Fujioka, Masato, Murayama, Ayako Y., Ogawa, Kaoru, Okano, Hideyuki, Ozawa, Hiroyuki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8307058/
https://www.ncbi.nlm.nih.gov/pubmed/34356098
http://dx.doi.org/10.3390/genes12071082
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author Hosoya, Makoto
Fujioka, Masato
Murayama, Ayako Y.
Ogawa, Kaoru
Okano, Hideyuki
Ozawa, Hiroyuki
author_facet Hosoya, Makoto
Fujioka, Masato
Murayama, Ayako Y.
Ogawa, Kaoru
Okano, Hideyuki
Ozawa, Hiroyuki
author_sort Hosoya, Makoto
collection PubMed
description Connexins are gap junction components that are essential for acquiring normal hearing ability. Up to 50% of congenital, autosomal-recessive, non-syndromic deafness can be attributed to variants in GJB2, the gene that encodes connexin 26. Gene therapies modifying the expression of connexins are a feasible treatment option for some patients with genetic hearing losses. However, the expression patterns of these proteins in the human fetus are not fully understood due to ethical concerns. Recently, the common marmoset was used as a primate animal model for the human fetus. In this study, we examined the expression patterns of connexin 26 and connexin 30 in the developing cochlea of this primate. Primate-specific spatiotemporal expression changes were revealed, which suggest the existence of primate-specific control of connexin expression patterns and specific functions of these gap junction proteins. Moreover, our results indicate that treatments for connexin-related hearing loss established in rodent models may not be appropriate for human patients, underscoring the importance of testing these treatments in primate models before applying them in human clinical trials.
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spelling pubmed-83070582021-07-25 Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea Hosoya, Makoto Fujioka, Masato Murayama, Ayako Y. Ogawa, Kaoru Okano, Hideyuki Ozawa, Hiroyuki Genes (Basel) Article Connexins are gap junction components that are essential for acquiring normal hearing ability. Up to 50% of congenital, autosomal-recessive, non-syndromic deafness can be attributed to variants in GJB2, the gene that encodes connexin 26. Gene therapies modifying the expression of connexins are a feasible treatment option for some patients with genetic hearing losses. However, the expression patterns of these proteins in the human fetus are not fully understood due to ethical concerns. Recently, the common marmoset was used as a primate animal model for the human fetus. In this study, we examined the expression patterns of connexin 26 and connexin 30 in the developing cochlea of this primate. Primate-specific spatiotemporal expression changes were revealed, which suggest the existence of primate-specific control of connexin expression patterns and specific functions of these gap junction proteins. Moreover, our results indicate that treatments for connexin-related hearing loss established in rodent models may not be appropriate for human patients, underscoring the importance of testing these treatments in primate models before applying them in human clinical trials. MDPI 2021-07-16 /pmc/articles/PMC8307058/ /pubmed/34356098 http://dx.doi.org/10.3390/genes12071082 Text en © 2021 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
Hosoya, Makoto
Fujioka, Masato
Murayama, Ayako Y.
Ogawa, Kaoru
Okano, Hideyuki
Ozawa, Hiroyuki
Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title_full Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title_fullStr Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title_full_unstemmed Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title_short Dynamic Spatiotemporal Expression Changes in Connexins of the Developing Primate’s Cochlea
title_sort dynamic spatiotemporal expression changes in connexins of the developing primate’s cochlea
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8307058/
https://www.ncbi.nlm.nih.gov/pubmed/34356098
http://dx.doi.org/10.3390/genes12071082
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