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ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms

Authors
 Kyeong Jee Cho  ;  Shin Hye Noh  ;  Soo Min Han  ;  Won-Il Choi  ;  Hye-Youn Kim  ;  Seyoung Yu  ;  Joon Suk Lee  ;  John Hoon Rim  ;  Min Goo Lee  ;  Friedhelm Hildebrandt  ;  Heon Yung Gee 
Citation
 PLOS GENETICS, Vol.14(3) : e1007316, 2018 
Journal Title
 PLOS GENETICS 
ISSN
 1553-7390 
Issue Date
2018
MeSH
Animals ; Axoneme/metabolism ; Cytoplasm/*metabolism ; DNA-Binding Proteins/genetics/*metabolism ; Dyneins/*metabolism ; Humans ; Mice ; Knockout Mice ; Phenotype ; Proteins/metabolism ; Messenger/metabolism RNA
Abstract
Zinc finger MYND-type-containing 10 (ZMYND10), a cytoplasmic protein expressed in ciliated cells, causes primary ciliary dyskinesia (PCD) when mutated; however, its function is poorly understood. Therefore, in this study, we examined the roles of ZMYND10 using Zmynd10-/-mice exhibiting typical PCD phenotypes, including hydrocephalus and laterality defects. In these mutants, morphology, the number of motile cilia, and the 9+2 axoneme structure were normal; however, inner and outer dynein arms (IDA and ODA, respectively) were absent. ZMYND10 interacted with ODA components and proteins, including LRRC6, DYX1C1, and C21ORF59, implicated in the cytoplasmic pre-assembly of DAs, whose levels were significantly reduced in Zmynd10-/-mice. LRRC6 and DNAI1 were more stable when co-expressed with ZYMND10 than when expressed alone. DNAI2, which did not interact with ZMYND10, was not stabilized by co-expression with ZMYND10 alone, but was stabilized by co-expression with DNAI1 and ZMYND10, suggesting that ZMYND10 stabilized DNAI1, which subsequently stabilized DNAI2. Together, these results demonstrated that ZMYND10 regulated the early stage of DA cytoplasmic pre-assembly by stabilizing DNAI1.
Files in This Item:
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DOI
10.1371/journal.pgen.1007316
Appears in Collections:
1. College of Medicine (의과대학) > Dept. of Pharmacology (약리학교실) > 1. Journal Papers
Yonsei Authors
Kim, Hye-Youn(김혜연) ORCID logo https://orcid.org/0000-0003-2090-6427
Noh, Shin Hye(노신혜)
Lee, Min Goo(이민구) ORCID logo https://orcid.org/0000-0001-7436-012X
Gee, Heon Yung(지헌영) ORCID logo https://orcid.org/0000-0002-8741-6177
Han, Soo Min(한수민)
URI
https://ir.ymlib.yonsei.ac.kr/handle/22282913/162350
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