PUBLICATION

Genomic non-redundancy of the mir-183/96/182 cluster and its requirement for hair cell maintenance

Authors
Fogerty, J., Stepanyan, R., Cianciolo, L.T., Tooke, B.P., Perkins, B.D.
ID
ZDB-PUB-190719-9
Date
2019
Source
Scientific Reports   9: 10302 (Journal)
Registered Authors
Fogerty, Joseph, Perkins, Brian
Keywords
none
MeSH Terms
  • Models, Animal
  • Animals
  • Hair Cells, Auditory/metabolism*
  • Gene Expression Profiling
  • Humans
  • Mutation
  • Multigene Family
  • Larva
  • Retina/metabolism*
  • MicroRNAs/genetics*
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Gene Expression Regulation, Developmental
(all 13)
PubMed
31311951 Full text @ Sci. Rep.
Abstract
microRNAs are important regulators of gene expression. In the retina, the mir-183/96/182 cluster is of particular interest due to its robust expression and studies in which loss of the cluster caused photoreceptor degeneration. However, it is unclear which of the three miRNAs in the cluster are ultimately required in photoreceptors, whether each may have independent, contributory roles, or whether a single miRNA from the cluster compensates for the loss of another. These are important questions that will not only help us to understand the role of these particular miRNAs in the retina, but will deepen our understanding of how clustered microRNAs evolve and operate. To that end, we have developed a complete panel of single, double, and triple mir-183/96/182 mutant zebrafish. While the retinas of all mutant animals were normal, the triple mutants exhibited acute hair cell degeneration which corresponded with impaired swimming and death at a young age. By measuring the penetrance of this phenotype in each mutant line, we determine which of the three miRNAs in the cluster are necessary and/or sufficient to ensure normal hair cell development and function.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
lri60
    Indel
    lri61
      Small Deletion
      lri69
        Deficiency
        lri70
          Indel
          lri78
            Deficiency
            lri79a
              Complex
              lri81
                Deficiency
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                Human Disease / Model
                No data available
                Sequence Targeting Reagents
                1 - 3 of 3
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                Fish
                Antibodies
                Orthology
                No data available
                Engineered Foreign Genes
                No data available
                Mapping
                No data available