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A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2019
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6614851/ https://www.ncbi.nlm.nih.gov/pubmed/31127312 http://dx.doi.org/10.1093/nar/gkz444 |
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author | Wang, Fei Ren, Dalong Liang, Xiaolin Ke, Shengwei Zhang, Bowen Hu, Bing Song, Xiaoyuan Wang, Xiangting |
author_facet | Wang, Fei Ren, Dalong Liang, Xiaolin Ke, Shengwei Zhang, Bowen Hu, Bing Song, Xiaoyuan Wang, Xiangting |
author_sort | Wang, Fei |
collection | PubMed |
description | Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the nonimprinting regions in brain (CNIBs). Among the seven identified CNIBs, human CNIB1 locus is located at Chr 9q33.3 and conserved from Danio rerio to Homo sapiens. Chr 9q33.3-9q34.11 microdeletion has previously been linked to human nail-patella syndrome (NPS) which is frequently accompanied by developmental and visual deficiencies. By generating CNIB1 deletion alleles in zebrafish, we demonstrated the requirement of CNIB1 for proper growth and development, and visual activities. Furthermore, we found that the role of CNIB1 on visual activity is mediated through a regulator of ocular development-lmx1bb. Collectively, our study shows that CNIB1 lncRNAs are important for zebrafish development and provides an lncRNA cluster-mediated pathophysiological mechanism for human Chr 9q33.3-9q34.11 microdeletion syndrome. |
format | Online Article Text |
id | pubmed-6614851 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-66148512019-07-12 A long noncoding RNA cluster-based genomic locus maintains proper development and visual function Wang, Fei Ren, Dalong Liang, Xiaolin Ke, Shengwei Zhang, Bowen Hu, Bing Song, Xiaoyuan Wang, Xiangting Nucleic Acids Res Genomics Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the nonimprinting regions in brain (CNIBs). Among the seven identified CNIBs, human CNIB1 locus is located at Chr 9q33.3 and conserved from Danio rerio to Homo sapiens. Chr 9q33.3-9q34.11 microdeletion has previously been linked to human nail-patella syndrome (NPS) which is frequently accompanied by developmental and visual deficiencies. By generating CNIB1 deletion alleles in zebrafish, we demonstrated the requirement of CNIB1 for proper growth and development, and visual activities. Furthermore, we found that the role of CNIB1 on visual activity is mediated through a regulator of ocular development-lmx1bb. Collectively, our study shows that CNIB1 lncRNAs are important for zebrafish development and provides an lncRNA cluster-mediated pathophysiological mechanism for human Chr 9q33.3-9q34.11 microdeletion syndrome. Oxford University Press 2019-07-09 2019-05-25 /pmc/articles/PMC6614851/ /pubmed/31127312 http://dx.doi.org/10.1093/nar/gkz444 Text en © The Author(s) 2019. Published by Oxford University Press on behalf of Nucleic Acids Research. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Genomics Wang, Fei Ren, Dalong Liang, Xiaolin Ke, Shengwei Zhang, Bowen Hu, Bing Song, Xiaoyuan Wang, Xiangting A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title | A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title_full | A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title_fullStr | A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title_full_unstemmed | A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title_short | A long noncoding RNA cluster-based genomic locus maintains proper development and visual function |
title_sort | long noncoding rna cluster-based genomic locus maintains proper development and visual function |
topic | Genomics |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6614851/ https://www.ncbi.nlm.nih.gov/pubmed/31127312 http://dx.doi.org/10.1093/nar/gkz444 |
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