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The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish
Chen, Jian-Yang1,2,3; Tan, Xungang4; Wang, Zheng-Hua1,2,3,4; Liu, Yun-Zhang1,2,3; Zhou, Jian-Feng1,2,3; Rong, Xiao-Zhi1,2,3; Lu, Ling1,2,3; Li, Yun1,2,3
2018-09-01
Source PublicationFEBS JOURNAL
ISSN1742-464X
Volume285Issue:18Pages:3464-3484
Corresponding AuthorLu, Ling(linglu@ouc.edu.cn) ; Li, Yun(yunlisun@ouc.edu.cn)
AbstractCraniofacial malformations are common congenital birth defects and usually caused by abnormal development of the cranial neural crest cells. Some nucleolar ribosome biogenesis factors are implicated in neural crest disorders also known as neurocristopathies. However, the underlying mechanisms linking ribosome biogenesis and neural crest cell (NCC) development remain to be elucidated. Here we report a novel zebrafish model with a CRISPR/Cas9-generated esf1 mutation, which exhibits severe NCC-derived pharyngeal cartilage loss and defects in the eyes, brain, and heart. The expression of several typical NCC markers, including sox10, dlx2a, nrp2b, crestin, vgll2a, and sox9a, was reduced in the head of the esf1 mutants, which indicates that esf1 plays a role in the development of zebrafish NCCs. We demonstrate that, similar to the yeast, loss of esf1 in zebrafish leads to defects in 18S rRNA biogenesis and ribosome biogenesis. We also show strong upregulation of p53 signaling as well as apoptosis, and poor proliferation in mutants. Inactivation of p53 rescues the early tissue defects and pharyngeal cartilage loss observed in esf1 mutants, indicating that increased cell death and pharyngeal cartilage defects observed in esf1 mutants are mediated via upregulated p53 signaling pathways. Based on transplantation analysis, we found esf1 functions in NCC in a cell autonomous fashion. Together, our results suggest that esf1 is required for NCC development and pharyngeal cartilage formation. These studies provide a potential model for investigating the relationship between ribosome biogenesis defects and craniofacial neurocristopathies.
Keywordapoptosis neural crest cells p53 pharyngeal cartilage
DOI10.1111/febs.14622
Indexed BySCI
Language英语
Funding ProjectNatural Scientific Foundation of China[31772457] ; Natural Scientific Foundation of China[31572261] ; Natural Scientific Foundation of China[31601863] ; Natural Science Foundation of Shandong Province, China[ZR2014CM007] ; NSFC-Shandong Joint Fund[U1406402] ; Scientific and Technological Innovation Project - Qingdao National Laboratory for Marine Science and Technology[2015ASKJ02]
WOS Research AreaBiochemistry & Molecular Biology
WOS SubjectBiochemistry & Molecular Biology
WOS IDWOS:000444682700010
PublisherWILEY
Citation statistics
Document Type期刊论文
Identifierhttp://ir.qdio.ac.cn/handle/337002/160218
Collection实验海洋生物学重点实验室
Corresponding AuthorLu, Ling; Li, Yun
Affiliation1.Ocean Univ China, Chinese Minist Educ, Key Lab Marine Drugs, 5 Yushan Rd, Qingdao 266003, Peoples R China
2.Ocean Univ China, Sch Med & Pharm, 5 Yushan Rd, Qingdao 266003, Peoples R China
3.Qingdao Natl Lab Marine Sci & Technol, Lab Marine Drugs & Biol Prod, Qingdao 266003, Peoples R China
4.Chinese Acad Sci, Inst Oceanol, CAS Key Lab Expt Marine Biol, Qingdao, Peoples R China
Recommended Citation
GB/T 7714
Chen, Jian-Yang,Tan, Xungang,Wang, Zheng-Hua,et al. The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish[J]. FEBS JOURNAL,2018,285(18):3464-3484.
APA Chen, Jian-Yang.,Tan, Xungang.,Wang, Zheng-Hua.,Liu, Yun-Zhang.,Zhou, Jian-Feng.,...&Li, Yun.(2018).The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish.FEBS JOURNAL,285(18),3464-3484.
MLA Chen, Jian-Yang,et al."The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish".FEBS JOURNAL 285.18(2018):3464-3484.
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