Primary cilia transduce diverse signals in embryonic development and adult tissues. Defective ciliogenesis results in a series of human disorders collectively known as ciliopathies. The CP110–CEP97 complex removal from the mother centriole is an early critical step for ciliogenesis, but the underlying mechanism for this step remains largely obscure. Here, we reveal that the linear ubiquitin chain assembly complex (LUBAC) plays an essential role in ciliogenesis by targeting the CP110–CEP97 complex. LUBAC specifically generates linear ubiquitin chains on CP110, which is required for CP110 removal from the mother centriole in ciliogenesis. We further identify that a pre-mRNA splicing factor, PRPF8, at the distal end of the mother centriole acts as the receptor of the linear ubiquitin chains to facilitate CP110 removal at the initial stage of ciliogenesis. Thus, our study reveals a direct mechanism of regulating CP110 removal in ciliogenesis and implicates the E3 ligase LUBAC as a potential therapy target of cilia-associated diseases, including ciliopathies and cancers.
Skip Nav Destination
Article navigation
3 January 2022
Article|
November 23 2021
LUBAC regulates ciliogenesis by promoting CP110 removal from the mother centriole
Xiao-Lin Shen,
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Jin-Feng Yuan,
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Xuan-He Qin,
Xuan-He Qin
3
School of Life Sciences and Technology, Tongji University, Shanghai, China
4
Shanghai East Hospital, Tongji University, Shanghai, China
Search for other works by this author on:
Guang-Ping Song,
Guang-Ping Song
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Huai-Bin Hu,
Huai-Bin Hu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Hai-Qing Tu,
Hai-Qing Tu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Zeng-Qing Song,
Zeng-Qing Song
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Pei-Yao Li,
Pei-Yao Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Yu-Ling Xu,
Yu-Ling Xu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Sen Li,
Sen Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Xiao-Xiao Jian,
Xiao-Xiao Jian
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Jia-Ning Li,
Jia-Ning Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Chun-Yu He,
Chun-Yu He
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Xi-Ping Yu,
Xi-Ping Yu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Li-Yun Liang,
Li-Yun Liang
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Min Wu,
Min Wu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Qiu-Ying Han,
Qiu-Ying Han
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Kai Wang,
Kai Wang
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Ai-Ling Li,
Ai-Ling Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Tao Zhou,
Tao Zhou
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Search for other works by this author on:
Yu-Cheng Zhang,
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Yu-Cheng Zhang: yczhang@xmail.ncba.ac.cn
Search for other works by this author on:
Na Wang,
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Na Wang: nwang@ncba.ac.cn
Search for other works by this author on:
Hui-Yan Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
2
School of Basic Medical Sciences, Fudan University, Shanghai, China
Correspondence to Hui-Yan Li: hyli@ncba.ac.cn
Search for other works by this author on:
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Xuan-He Qin
3
School of Life Sciences and Technology, Tongji University, Shanghai, China
4
Shanghai East Hospital, Tongji University, Shanghai, China
Guang-Ping Song
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Huai-Bin Hu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Hai-Qing Tu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Zeng-Qing Song
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Pei-Yao Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Yu-Ling Xu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Sen Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Xiao-Xiao Jian
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Jia-Ning Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Chun-Yu He
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Xi-Ping Yu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Li-Yun Liang
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Min Wu
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Qiu-Ying Han
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Kai Wang
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Ai-Ling Li
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Tao Zhou
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
1
Nanhu Laboratory, State Key Laboratory of Proteomics, National Center of Biomedical Analysis, Beijing, China
Correspondence to Hui-Yan Li: hyli@ncba.ac.cn
Na Wang: nwang@ncba.ac.cn
Yu-Cheng Zhang: yczhang@xmail.ncba.ac.cn
*
X.-L. Shen and J.-F. Yuan contributed equally to this paper.
Received:
May 16 2021
Revision Received:
September 13 2021
Accepted:
October 15 2021
Online Issn: 1540-8140
Print Issn: 0021-9525
Funding
Funder(s):
National Basic Research Program of China
- Award Id(s): 2014CB910603
Funder(s):
National Natural Science Foundation of China
- Award Id(s): 81521064,81790252,82002970
Funder(s):
National Key Research and Development Program
- Award Id(s): 2017YFC1601100,2017YFC1601101,2017YFC1601102,2017YFC1601104,2017YFA0503900
© 2021 Shen et al.
2021
This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see http://www.rupress.org/terms/). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 4.0 International license, as described at https://creativecommons.org/licenses/by-nc-sa/4.0/).
J Cell Biol (2022) 221 (1): e202105092.
Article history
Received:
May 16 2021
Revision Received:
September 13 2021
Accepted:
October 15 2021
Citation
Xiao-Lin Shen, Jin-Feng Yuan, Xuan-He Qin, Guang-Ping Song, Huai-Bin Hu, Hai-Qing Tu, Zeng-Qing Song, Pei-Yao Li, Yu-Ling Xu, Sen Li, Xiao-Xiao Jian, Jia-Ning Li, Chun-Yu He, Xi-Ping Yu, Li-Yun Liang, Min Wu, Qiu-Ying Han, Kai Wang, Ai-Ling Li, Tao Zhou, Yu-Cheng Zhang, Na Wang, Hui-Yan Li; LUBAC regulates ciliogenesis by promoting CP110 removal from the mother centriole. J Cell Biol 3 January 2022; 221 (1): e202105092. doi: https://doi.org/10.1083/jcb.202105092
Download citation file:
Sign in
Don't already have an account? Register
Client Account
You could not be signed in. Please check your email address / username and password and try again.
Sign in via your Institution
Sign in via your Institution
1,182
Views
0
Citations
Advertisement
Advertisement