The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish

doi:10.1111/febs.14622

CORC > 海洋研究所 > 中国科学院海洋研究所 > 实验海洋生物学重点实验室

	The ribosome biogenesis protein Esf1 is essential for pharyngeal cartilage formation in zebrafish
	Chen, Jian-Yang 1,2,3; Tan, Xungang4 ; Wang, Zheng-Hua 1,2,3,4; Liu, Yun-Zhang 1,2,3; Zhou, Jian-Feng 1,2,3; Rong, Xiao-Zhi 1,2,3; Lu, Ling 1,2,3; Li, Yun 1,2,3
刊名	FEBS JOURNAL
	2018-09-01
卷号	285 期号:18 页码:3464-3484
关键词	apoptosis neural crest cells p53 pharyngeal cartilage
ISSN号	1742-464X
DOI	10.1111/febs.14622
通讯作者	Lu, Ling(linglu@ouc.edu.cn) ; Li, Yun(yunlisun@ouc.edu.cn)
英文摘要	Craniofacial 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.
资助项目	Natural 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研究方向	Biochemistry & Molecular Biology
语种	英语
出版者	WILEY
WOS记录号	WOS:000444682700010
内容类型	期刊论文
源URL	[http://ir.qdio.ac.cn/handle/337002/160217]
专题	海洋研究所_实验海洋生物学重点实验室
通讯作者	Lu, Ling; Li, Yun
作者单位	1.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
推荐引用方式 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.