PUBLICATION

The zebrafish dog-eared mutation disrupts eya1, a gene required for cell survival and differentiation in the inner ear and lateral line

Authors
Kozlowski, D.J., Whitfield, T.T., Hukriede, N.A., Lam, W.K., and Weinberg, E.S.
ID
ZDB-PUB-041202-4
Date
2005
Source
Developmental Biology   277(1): 27-41 (Journal)
Registered Authors
Hukriede, Neil, Kozlowski, David J., Weinberg, Eric, Whitfield, Tanya T.
Keywords
eya1; Otic vesicle; Cristae; Fate mapping; Zebrafish; bmp4
MeSH Terms
  • Hair Cells, Vestibular/embryology
  • Chromosome Mapping
  • Bone Morphogenetic Protein 4
  • Mutation
  • Animals
  • Ear, Inner/embryology*
  • Trans-Activators/genetics
  • Trans-Activators/physiology*
  • Zebrafish
  • Apoptosis
  • RNA, Messenger/analysis
  • Protein Tyrosine Phosphatases
  • Bone Morphogenetic Proteins/genetics
  • Zebrafish Proteins
  • Nuclear Proteins
  • Intracellular Signaling Peptides and Proteins
(all 16)
PubMed
15572137 Full text @ Dev. Biol.
Abstract
To understand the molecular basis of sensory organ development and disease, we have cloned and characterized the zebrafish mutation dog-eared (dog) that is defective in formation of the inner ear and lateral line sensory systems. The dog locus encodes the eyes absent-1 (eya1) gene and single point mutations were found in three independent dog alleles, each prematurely truncating the expressed protein within the Eya domain. Moreover, morpholino-mediated knockdown of eya1 gene function phenocopies the dog-eared mutation. In zebrafish, the eya1 gene is widely expressed in placode-derived sensory organs during embryogenesis but Eya1 function appears to be primarily required for survival of sensory hair cells in the developing ear and lateral line neuromasts. Increased levels of apoptosis occur in the migrating primordia of the posterior lateral line in dog embryos and as well as in regions of the developing otocyst that are mainly fated to give rise to sensory cells of the cristae. Importantly, mutation of the EYA1 or EYA4 gene causes hereditary syndromic deafness in humans. Determination of eya gene function during zebrafish organogenesis will facilitate understanding the molecular etiology of human vestibular and hearing disorders.
Genes / Markers
Figures
Figure Gallery (9 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
eya1tc257e/tc257estandard conditionsPrim-5
eya1tc257e/tc257estandard conditionsLong-pec
eya1tc257e/tc257estandard conditionsLong-pec
eya1tc257e/tc257estandard conditionsDay 4
eya1tc257e/tc257estandard conditionsDay 4
eya1tc257e/tc257estandard conditionsDays 7-13
eya1tc257e/tc257estandard conditionsDays 7-13
eya1tc257e/tc257estandard conditionsDays 7-13
eya1tm90b/tm90bstandard conditionsPrim-5
eya1tm90b/tm90bstandard conditionsPrim-5
1 - 10 of 39
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
tc257e
    		Point Mutation
    tm90b
      		Point Mutation
      tp85b
        		Point Mutation
        1 - 3 of 3
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        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        eya1MO1-eya1MRPHLNO
        1 - 1 of 1
        Show
        Fish
        Fish
        eya1tc257e/tc257e
        eya1tm90b/tm90b
        eya1tp85b/tp85b
        WT
        1 - 4 of 4
        Show
        Antibodies
        No data available
        Orthology
        No data available
        Engineered Foreign Genes
        No data available
        Mapping
        Entity Type Entity Symbol Location
        ESTfc13c10Chr: 24 Details
        Featuretm90bChr: 24 Details
        SSLPz7823Chr: 24 Details
        SSLPz9321Chr: 24 Details
        1 - 4 of 4
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        Citation
        Kozlowski, D.J., Whitfield, T.T., Hukriede, N.A., Lam, W.K., and Weinberg, E.S. (2005) The zebrafish dog-eared mutation disrupts eya1, a gene required for cell survival and differentiation in the inner ear and lateral line. Developmental Biology. 277(1):27-41.