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Multiple Developmental Defects in sox11a Mutant Zebrafish with Features of Coffin-Siris Syndrome

文献类型：期刊论文

作者Jia, Shaoting1,3; Wu, Xingxing1,3; Wu, Yunya1,3; Cui, Xuefan1,3; Tao, Binbin1,3; Zhu, Zuoyan3; Hu, Wei1,2,3
刊名INTERNATIONAL JOURNAL OF BIOLOGICAL SCIENCES
出版日期2020
卷号16期号:15页码:3039-3049
ISSN号1449-2288
关键词sox11 zebrafish Coffin-Siris CRISPR/Cas9
DOI10.7150/ijbs.47510
通讯作者Hu, Wei(huwei@ihb.ac.cn)
英文摘要A previous study suggested that human Coffin-Siris syndrome is related to the mutation of SOX11. Since the homozygous SOX11 mutant mice died soon after birth, no suitable model was available for the study of the pathogenic mechanism of Coffin-Siris syndrome. To solve this problem, we generated two viable homozygous zebrafish mutants, sox11a(m/m) and sox11b(m/m). We found that the sox11a(m/m) mutant possessed Coffin-Siris syndrome features. The sox11a(m/m) mutants exhibited growth deficiency from 3.3 hpf embryos to adulthood. Furthermore, the sox11a(m/m) mutant also displayed microcephaly, narrow pupillary distance, achondroplasia, and bone deformity in adults. Growth deficiency could be rescued by the injection of sox11a mRNA at the one-cell stage. In addition, the expression levels of genes related to cartilage and bone were downregulated in the sox11a(m/m) mutant, indicating that sox11a mainly affected the growth and development of zebrafish by regulating the expression of genes related to skeletal development. Our results indicate that sox11a(m/m) mutant zebrafish offered a potential model system to help with the search for pathogenic mechanisms of human Coffin-Siris syndrome.
WOS关键词GENE ; SPECIFICATION ; SEC23A
资助项目National Key R&D Program of China[2018YFA0801000] ; National Natural Science Foundation of China[31721005] ; Chinese Academy of Sciences[2019FBZ05]
WOS研究方向Biochemistry & Molecular Biology ; Life Sciences & Biomedicine - Other Topics
语种英语
出版者IVYSPRING INT PUBL
WOS记录号WOS:000585302400001
资助机构National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Key R&D Program of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences
源URL[http://ir.ihb.ac.cn/handle/342005/39269]  
专题水生生物研究所_鱼类生物学及渔业生物技术研究中心_期刊论文
通讯作者Hu, Wei
作者单位1.Univ Chinese Acad Sci, Beijing 100049, Peoples R China
2.Qingdao Natl Lab Marine Sci & Technol, Qingdao 266237, Peoples R China
3.Chinese Acad Sci, Inst Hydrobiol, State Key Lab Freshwater Ecol & Biotechnol, Wuhan 430072, Peoples R China
推荐引用方式
GB/T 7714		 Jia, Shaoting,Wu, Xingxing,Wu, Yunya,et al. Multiple Developmental Defects in sox11a Mutant Zebrafish with Features of Coffin-Siris Syndrome[J]. INTERNATIONAL JOURNAL OF BIOLOGICAL SCIENCES,2020,16(15):3039-3049.
APA Jia, Shaoting.,Wu, Xingxing.,Wu, Yunya.,Cui, Xuefan.,Tao, Binbin.,...&Hu, Wei.(2020).Multiple Developmental Defects in sox11a Mutant Zebrafish with Features of Coffin-Siris Syndrome.INTERNATIONAL JOURNAL OF BIOLOGICAL SCIENCES,16(15),3039-3049.
MLA Jia, Shaoting,et al."Multiple Developmental Defects in sox11a Mutant Zebrafish with Features of Coffin-Siris Syndrome".INTERNATIONAL JOURNAL OF BIOLOGICAL SCIENCES 16.15(2020):3039-3049.

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来源：水生生物研究所

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