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IHB OpenIR  > 鱼类生物学及渔业生物技术研究中心  > 期刊论文
Multiple Developmental Defects in sox11a Mutant Zebrafish with Features of Coffin-Siris Syndrome
Jia, Shaoting1,3; Wu, Xingxing1,3; Wu, Yunya1,3; Cui, Xuefan1,3; Tao, Binbin1,3; Zhu, Zuoyan1; Hu, Wei1,2,3
Corresponding AuthorHu, Wei(huwei@ihb.ac.cn)
2020
Source PublicationINTERNATIONAL JOURNAL OF BIOLOGICAL SCIENCES
ISSN1449-2288
Volume16Issue:15Pages:3039-3049
AbstractA 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.
Keywordsox11 zebrafish Coffin-Siris CRISPR/Cas9
DOI10.7150/ijbs.47510
Funding OrganizationNational 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
Indexed BySCI ; SCI
Language英语
Funding ProjectNational Key R&D Program of China[2018YFA0801000] ; National Natural Science Foundation of China[31721005] ; Chinese Academy of Sciences[2019FBZ05]
WOS Research AreaBiochemistry & Molecular Biology ; Life Sciences & Biomedicine - Other Topics
WOS SubjectBiochemistry & Molecular Biology ; Biology
WOS IDWOS:000585302400001
WOS KeywordGENE ; SPECIFICATION ; SEC23A
PublisherIVYSPRING INT PUBL
Funding OrganizationNational 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
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Document Type期刊论文
Identifierhttp://ir.ihb.ac.cn/handle/342005/39270
Collection鱼类生物学及渔业生物技术研究中心_期刊论文
Corresponding AuthorHu, Wei
Affiliation1.Chinese Acad Sci, Inst Hydrobiol, State Key Lab Freshwater Ecol & Biotechnol, Wuhan 430072, Peoples R China
2.Qingdao Natl Lab Marine Sci & Technol, Qingdao 266237, Peoples R China
3.Univ Chinese Acad Sci, Beijing 100049, Peoples R China
Recommended Citation
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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