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Hyccin/FAM126A deficiency reduces glial enrichment and axonal sheath, which are rescued by overexpression of a plasma membrane- targeting PI4KIIIa in Drosophila

文献类型：期刊论文

作者Zhang, Qichao2,3,4,5; Zhang, Baozhu6,11; Lim, Nastasia K. H.1,4,7; Zhang, Xiao6,11; Meng, Shiquan4,8; Nyengaard, Jens R.2,3,5; Huang, Fude1,2,3,4; Wang, Wen-An9,10
刊名BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS
出版日期2022-01-22
卷号589页码:71-77
ISSN号0006-291X
关键词Glia FAM126A FAM126B PI4KIII a Hyccin TTC7 Phosphatidylinositols
DOI10.1016/j.bbrc.2021.11.106
通讯作者Huang, Fude(huangfude69@163.com) ; Wang, Wen-An(wangwenan@xinhuamed.com.cn)
英文摘要Hyccin/FAM126A mutations are linked to hypomyelination and congenital cataract disease (HCC), but whether and how Hyccin/FAM126A deficiency causes hypomyelination remains undetermined. This study shows Hyccin/FAM126A expression was necessary for the expression of other components of the PI4KIIIa complex in Drosophila. Knockdown of Hyccin/FAM126A in glia reduced the enrichment of glial cells, disrupted axonal sheaths and visual ability in the visual system, and these defects could be fully rescued by overexpressing either human FAM126A or FAM126B, and partially rescued by overexpressing a plasma membrane-targeting recombinant mouse PI4KIIIa. Additionally, PI4KIIIa knockdown in glia phenocopied Hyccin/FAM126A knockdown, and this was partially rescued by overexpressing the recombinant PI4KIIIa, but not human FAM126A or FAM126B. This study establishes an animal model of HCC and indicates that Hyccin/FAM126A plays an essential role in glial enrichment and axonal sheath in a cell-autonomous manner in the visual system via controlling the expression and stabilization of the PI4KIIIa complex at the plasma membrane. (c) 2021 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
WOS关键词HYPOMYELINATION ; PROTEIN ; HYCCIN
资助项目National Natural Science Foundation of China[81650110527] ; National Natural Science Foundation of China[81771416] ; National Natural Science Foundation of China[81971000] ; National Natural Science Foundation of China[81371400] ; National Key Basic Research Program of China[2013CB530900]
WOS研究方向Biochemistry & Molecular Biology ; Biophysics
语种英语
出版者ACADEMIC PRESS INC ELSEVIER SCIENCE
WOS记录号WOS:000733564600006
源URL[http://119.78.100.183/handle/2S10ELR8/300815]  
专题中国科学院上海药物研究所
通讯作者Huang, Fude; Wang, Wen-An
作者单位1.Nuobeta Pharmaceut Technol Shanghai Co Ltd, Shanghai 201210, Peoples R China
2.Univ Chinese Acad Sci, Sino Danish Coll, Beijing 100190, Peoples R China
3.Sino Danish Ctr Educ & Res, Beijing 100190, Peoples R China
4.Chinese Acad Sci, Univ Chinese Acad Sci, Shanghai Adv Res Inst, Shanghai 201210, Peoples R China
5.Aarhus Univ, Core Ctr Mol Morphol, Dept Clin Med, Sect Stereol & Microscopy, DK-8200 Aarhus, Denmark
6.Chinese Acad Sci, State Key Lab Neurosci, Shanghai Inst Biol Sci, Shanghai 200031, Peoples R China
7.Chinese Acad Sci, Univ Chinese Acad Sci, Shanghai Inst Mat Med, Shanghai 201203, Peoples R China
8.Shanghai Univ, Sch Life Sci, Lab Mol Neurobiol, Shanghai 200072, Peoples R China
9.Shanghai Jiao Tong Univ, Sch Med, Xinhua Hosp, Dept Neurol, Shanghai 200092, Peoples R China
10.Xinhua Hosp, Dept Neurol, Chongming Branch, Shanghai 202150, Peoples R China
推荐引用方式
GB/T 7714		 Zhang, Qichao,Zhang, Baozhu,Lim, Nastasia K. H.,et al. Hyccin/FAM126A deficiency reduces glial enrichment and axonal sheath, which are rescued by overexpression of a plasma membrane- targeting PI4KIIIa in Drosophila[J]. BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS,2022,589:71-77.
APA Zhang, Qichao.,Zhang, Baozhu.,Lim, Nastasia K. H..,Zhang, Xiao.,Meng, Shiquan.,...&Wang, Wen-An.(2022).Hyccin/FAM126A deficiency reduces glial enrichment and axonal sheath, which are rescued by overexpression of a plasma membrane- targeting PI4KIIIa in Drosophila.BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS,589,71-77.
MLA Zhang, Qichao,et al."Hyccin/FAM126A deficiency reduces glial enrichment and axonal sheath, which are rescued by overexpression of a plasma membrane- targeting PI4KIIIa in Drosophila".BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS 589(2022):71-77.

入库方式： OAI收割

来源：上海药物研究所

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