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斑马鱼通过调节Rho-GTPase信号通路影响先天性听力。

Zebrafish affects congenital hearing by regulating Rho-GTPase signaling.

作者信息

Xie Binling, Liang Jiaxin, Jiang Jifan, Zeng Ting, Liu Ling, Xie Dinghua, Zhu Ganghua, Xiong Lei, Zhang Kanjia, Liu Dong, Gong Jie, Chen Xiangding, Lai Ruosha, Xie Huaping

机构信息

Laboratory of Animal Nutrition and Human Health, Hunan International Joint Laboratory of Animal Intestinal Ecology and Health, College of Life Science, Hunan Normal University, Changsha, Hunan, China.

State Key Laboratory of Developmental Biology of Freshwater Fish, College of Life Science, Hunan Normal University, Changsha, Hunan, China.

出版信息

Front Mol Neurosci. 2024 Jul 15;17:1405109. doi: 10.3389/fnmol.2024.1405109. eCollection 2024.

DOI:10.3389/fnmol.2024.1405109
PMID:39081296
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11287254/
Abstract

INTRODUCTION

, the homolog of the human Usher 1B syndrome pathogenic gene, , plays an important role in stereociliary development and maintenance, therefore, is critical for hearing and balance. However, the molecular mechanisms that regulate hearing and balance still need to be studied.

METHODS

In this study, we generated two independent zebrafish knockout lines using CRISPR/Cas9 technology. To investigate the effects of on hearing, YO-PRO-1 staining and startle response assay were used. To gain insight into the specific molecular mechanisms by which affects hearing, transcriptome sequencing and bioinformatics analysis were employed.

RESULTS

Our study showed that hair cells of -/- zebrafish can not take up YO-PRO-1 fluorescent dye and are insensitive to acoustic stimulation in -/- zebrafish compared to wild type. Genes related to the Rho GTPase signaling pathway, such as arhgap33, dab2ip, and arghef40, are significantly down-regulated in -/- zebrafish embryos at 3 dpf. GTP and ATP compensation can partially rescue the hair cell defects in knockout zebrafish.

DISCUSSION

Our findings suggest that zebrafish affects congenital hearing by regulating Rho GTPase signaling, and loss of leads to abnormal Rho GTPase signaling and impairs hair cell function. , , arhgap33, dab2ip, arghef40 and -/- fonts in the abstract are italicized. -/- is a superscript format.

摘要

引言

人类乌谢尔综合征1B型致病基因的同源物，在静纤毛的发育和维持中起重要作用，因此对听力和平衡至关重要。然而，调节听力和平衡的分子机制仍有待研究。

方法

在本研究中，我们使用CRISPR/Cas9技术生成了两个独立的斑马鱼基因敲除品系。为了研究该基因对听力的影响，采用了YO-PRO-1染色和惊吓反应试验。为了深入了解该基因影响听力的具体分子机制，进行了转录组测序和生物信息学分析。

结果

我们的研究表明，与野生型相比，该基因敲除的斑马鱼的毛细胞不能摄取YO-PRO-1荧光染料，并且对声音刺激不敏感。在3天胚胎期的该基因敲除斑马鱼胚胎中，与Rho GTPase信号通路相关的基因，如arhgap33、dab2ip和arghef40，显著下调。GTP和ATP补偿可以部分挽救该基因敲除斑马鱼的毛细胞缺陷。

讨论

我们的研究结果表明，斑马鱼该基因通过调节Rho GTPase信号通路影响先天性听力，该基因的缺失导致Rho GTPase信号异常并损害毛细胞功能。摘要中的该基因、arhgap33、dabip、arghef40和基因敲除为斜体格式。基因敲除为上标格式。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/db3bb5b34168/fnmol-17-1405109-g008.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/417d96d7074e/fnmol-17-1405109-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/9aed62edbc19/fnmol-17-1405109-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/f88dda14db69/fnmol-17-1405109-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/d15f35206e88/fnmol-17-1405109-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/ba745c7ce900/fnmol-17-1405109-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/0a10f0363fe6/fnmol-17-1405109-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/f0e9a7b2770c/fnmol-17-1405109-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/db3bb5b34168/fnmol-17-1405109-g008.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/417d96d7074e/fnmol-17-1405109-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/9aed62edbc19/fnmol-17-1405109-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/f88dda14db69/fnmol-17-1405109-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/d15f35206e88/fnmol-17-1405109-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/ba745c7ce900/fnmol-17-1405109-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/0a10f0363fe6/fnmol-17-1405109-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/f0e9a7b2770c/fnmol-17-1405109-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1765/11287254/db3bb5b34168/fnmol-17-1405109-g008.jpg

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