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ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms
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...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2018
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5895051/ https://www.ncbi.nlm.nih.gov/pubmed/29601588 http://dx.doi.org/10.1371/journal.pgen.1007316 |
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author | Cho, Kyeong Jee Noh, Shin Hye Han, Soo Min Choi, Won-Il Kim, Hye-Youn Yu, Seyoung Lee, Joon Suk Rim, John Hoon Lee, Min Goo Hildebrandt, Friedhelm Gee, Heon Yung |
author_facet | Cho, Kyeong Jee Noh, Shin Hye Han, Soo Min Choi, Won-Il Kim, Hye-Youn Yu, Seyoung Lee, Joon Suk Rim, John Hoon Lee, Min Goo Hildebrandt, Friedhelm Gee, Heon Yung |
author_sort | Cho, Kyeong Jee |
collection | PubMed |
description | 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. |
format | Online Article Text |
id | pubmed-5895051 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2018 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
spelling | pubmed-58950512018-05-04 ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms Cho, Kyeong Jee Noh, Shin Hye Han, Soo Min Choi, Won-Il Kim, Hye-Youn Yu, Seyoung Lee, Joon Suk Rim, John Hoon Lee, Min Goo Hildebrandt, Friedhelm Gee, Heon Yung PLoS Genet Research Article 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. Public Library of Science 2018-03-30 /pmc/articles/PMC5895051/ /pubmed/29601588 http://dx.doi.org/10.1371/journal.pgen.1007316 Text en © 2018 Cho et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
spellingShingle | Research Article Cho, Kyeong Jee Noh, Shin Hye Han, Soo Min Choi, Won-Il Kim, Hye-Youn Yu, Seyoung Lee, Joon Suk Rim, John Hoon Lee, Min Goo Hildebrandt, Friedhelm Gee, Heon Yung ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title | ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title_full | ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title_fullStr | ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title_full_unstemmed | ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title_short | ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
title_sort | zmynd10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5895051/ https://www.ncbi.nlm.nih.gov/pubmed/29601588 http://dx.doi.org/10.1371/journal.pgen.1007316 |
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