Molecular mechanisms underlying the role of the centriolar CEP164-TTBK2 complex in ciliopathies
The result's identifiers
Result code in IS VaVaI
<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00216224%3A14110%2F22%3A00125148" target="_blank" >RIV/00216224:14110/22:00125148 - isvavai.cz</a>
Result on the web
<a href="https://www.sciencedirect.com/science/article/pii/S0969212621003026" target="_blank" >https://www.sciencedirect.com/science/article/pii/S0969212621003026</a>
DOI - Digital Object Identifier
<a href="http://dx.doi.org/10.1016/j.str.2021.08.007" target="_blank" >10.1016/j.str.2021.08.007</a>
Alternative languages
Result language
angličtina
Original language name
Molecular mechanisms underlying the role of the centriolar CEP164-TTBK2 complex in ciliopathies
Original language description
Cilia formation is essential for human life. One of the earliest events in the ciliogenesis program is the recruitment of tau-tubulin kinase 2 (TTBK2) by the centriole distal appendage component CEP164. Due to the lack of high-resolution structural information on this complex, it is unclear how it is affected in human ciliopathies such as nephronophthisis. Furthermore, it is poorly understood if binding to CEP164 influences TTBK2 activities. Here, we present a detailed biochemical, structural, and functional analysis of the CEP164-TTBK2 complex and demonstrate how it is compromised by two ciliopathic mutations in CEP164. Moreover, we also provide insights into how binding to CEP164 is coordinated with TTBK2 activities. Together, our data deepen our understanding of a crucial step in cilia formation and will inform future studies aimed at restoring CEP164 functionality in a debilitating human ciliopathy.
Czech name
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Czech description
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Classification
Type
J<sub>imp</sub> - Article in a specialist periodical, which is included in the Web of Science database
CEP classification
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OECD FORD branch
10601 - Cell biology
Result continuities
Project
Result was created during the realization of more than one project. More information in the Projects tab.
Continuities
P - Projekt vyzkumu a vyvoje financovany z verejnych zdroju (s odkazem do CEP)
Others
Publication year
2022
Confidentiality
S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů
Data specific for result type
Name of the periodical
Structure
ISSN
0969-2126
e-ISSN
1878-4186
Volume of the periodical
30
Issue of the periodical within the volume
1
Country of publishing house
US - UNITED STATES
Number of pages
25
Pages from-to
„114-128“-„e1-e9“
UT code for WoS article
000742422300013
EID of the result in the Scopus database
2-s2.0-85120778742