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Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function
Piccolo is a presynaptic protein with high conservation among different species, and the expression of Piccolo is extensive in vertebrates. Recently, a small fragment of Piccolo (Piccolino), arising due to the incomplete splicing of intron 5/6, was found to be present in the synapses of retinas and...
Autores principales: | , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Impact Journals
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8109093/ https://www.ncbi.nlm.nih.gov/pubmed/33882456 http://dx.doi.org/10.18632/aging.202861 |
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author | Li, Peipei Lin, Zhuchun An, Yachun Lin, Jing Zhang, Aizhen Wang, Shuangyan Tu, Hailong Ran, Jie Wang, Jinpeng Liang, Yu Liu, Ziyi Ye, Chao Fu, Xiaolong Gao, Jiangang |
author_facet | Li, Peipei Lin, Zhuchun An, Yachun Lin, Jing Zhang, Aizhen Wang, Shuangyan Tu, Hailong Ran, Jie Wang, Jinpeng Liang, Yu Liu, Ziyi Ye, Chao Fu, Xiaolong Gao, Jiangang |
author_sort | Li, Peipei |
collection | PubMed |
description | Piccolo is a presynaptic protein with high conservation among different species, and the expression of Piccolo is extensive in vertebrates. Recently, a small fragment of Piccolo (Piccolino), arising due to the incomplete splicing of intron 5/6, was found to be present in the synapses of retinas and cochleae. However, the comprehensive function of Piccolo in the retina and cochlea remains unclear. In this study, we generated Piccolo knockout mice using CRISPR-Cas9 technology to explore the function of Piccolo. Unexpectedly, whereas no abnormalities were found in the cochlear hair cells of the mutant mice, significant differences were found in the retinas, in which two layers (the outer nuclear layer and the outer plexiform layer) were absent. Additionally, the amplitudes of electroretinograms were significantly reduced and pigmentation was observed in the fundoscopy of the mutant mouse retinas. The expression levels of Bassoon, a homolog of Piccolo, as well as synapse-associated proteins CtBP1, CtBP2, Kif3A, and Rim1 were down-regulated. The numbers of ribbon synapses in the retinas of the mutant mice were also reduced. Altogether, the phenotype of Piccolo-/- mice resembled the symptoms of retinitis pigmentosa (RP) in humans, suggesting Piccolo might be a candidate gene of RP and indicates Piccolo knockout mice are a good model for elucidating the molecular mechanisms of RP. |
format | Online Article Text |
id | pubmed-8109093 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Impact Journals |
record_format | MEDLINE/PubMed |
spelling | pubmed-81090932021-05-12 Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function Li, Peipei Lin, Zhuchun An, Yachun Lin, Jing Zhang, Aizhen Wang, Shuangyan Tu, Hailong Ran, Jie Wang, Jinpeng Liang, Yu Liu, Ziyi Ye, Chao Fu, Xiaolong Gao, Jiangang Aging (Albany NY) Research Paper Piccolo is a presynaptic protein with high conservation among different species, and the expression of Piccolo is extensive in vertebrates. Recently, a small fragment of Piccolo (Piccolino), arising due to the incomplete splicing of intron 5/6, was found to be present in the synapses of retinas and cochleae. However, the comprehensive function of Piccolo in the retina and cochlea remains unclear. In this study, we generated Piccolo knockout mice using CRISPR-Cas9 technology to explore the function of Piccolo. Unexpectedly, whereas no abnormalities were found in the cochlear hair cells of the mutant mice, significant differences were found in the retinas, in which two layers (the outer nuclear layer and the outer plexiform layer) were absent. Additionally, the amplitudes of electroretinograms were significantly reduced and pigmentation was observed in the fundoscopy of the mutant mouse retinas. The expression levels of Bassoon, a homolog of Piccolo, as well as synapse-associated proteins CtBP1, CtBP2, Kif3A, and Rim1 were down-regulated. The numbers of ribbon synapses in the retinas of the mutant mice were also reduced. Altogether, the phenotype of Piccolo-/- mice resembled the symptoms of retinitis pigmentosa (RP) in humans, suggesting Piccolo might be a candidate gene of RP and indicates Piccolo knockout mice are a good model for elucidating the molecular mechanisms of RP. Impact Journals 2021-04-21 /pmc/articles/PMC8109093/ /pubmed/33882456 http://dx.doi.org/10.18632/aging.202861 Text en Copyright: © 2021 Li et al. https://creativecommons.org/licenses/by/3.0/This is an open access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/3.0/) (CC BY 3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
spellingShingle | Research Paper Li, Peipei Lin, Zhuchun An, Yachun Lin, Jing Zhang, Aizhen Wang, Shuangyan Tu, Hailong Ran, Jie Wang, Jinpeng Liang, Yu Liu, Ziyi Ye, Chao Fu, Xiaolong Gao, Jiangang Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title | Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title_full | Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title_fullStr | Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title_full_unstemmed | Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title_short | Piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
title_sort | piccolo is essential for the maintenance of mouse retina but not cochlear hair cell function |
topic | Research Paper |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8109093/ https://www.ncbi.nlm.nih.gov/pubmed/33882456 http://dx.doi.org/10.18632/aging.202861 |
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