PUBLICATION

Kremen1 regulates mechanosensory hair cell development in the mammalian cochlea and the zebrafish lateral line

Authors
Mulvaney, J.F., Thompkins, C., Noda, T., Nishimura, K., Sun, W.W., Lin, S.Y., Coffin, A., Dabdoub, A.
ID
ZDB-PUB-160824-4
Date
2016
Source
Scientific Reports   6: 31668 (Journal)
Registered Authors
Coffin, Allison, Lin, Shuh-Yow
Keywords
Cochlea, Organogenesis
MeSH Terms
  • Animals
  • Animals, Genetically Modified
  • Cell Differentiation/genetics
  • Cochlea/embryology
  • Cochlea/growth & development
  • Cochlea/metabolism*
  • Gene Expression Regulation, Developmental*
  • Hair Cells, Auditory/metabolism*
  • Lateral Line System/embryology
  • Lateral Line System/growth & development
  • Lateral Line System/metabolism*
  • Mechanoreceptors/metabolism
  • Membrane Proteins/genetics*
  • Membrane Proteins/metabolism
  • Mice
  • Mutation
  • Neurogenesis/genetics
  • RNA Interference
  • Zebrafish
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
(all 21)
PubMed
27550540 Full text @ Sci. Rep.
Abstract
Here we present spatio-temporal localization of Kremen1, a transmembrane receptor, in the mammalian cochlea, and investigate its role in the formation of sensory organs in mammal and fish model organisms. We show that Kremen1 is expressed in prosensory cells during cochlear development and in supporting cells of the adult mouse cochlea. Based on this expression pattern, we investigated whether Kremen1 functions to modulate cell fate decisions in the prosensory domain of the developing cochlea. We used gain and loss-of-function experiments to show that Kremen1 is sufficient to bias cells towards supporting cell fate, and is implicated in suppression of hair cell formation. In addition to our findings in the mouse cochlea, we examined the effects of over expression and loss of Kremen1 in the zebrafish lateral line. In agreement with our mouse data, we show that over expression of Kremen1 has a negative effect on the number of mechanosensory cells that form in the zebrafish neuromasts, and that fish lacking Kremen1 protein develop more hair cells per neuromast compared to wild type fish. Collectively, these data support an inhibitory role for Kremen1 in hair cell fate specification.
Genes / Markers
Marker Marker Type Name
kremen1GENEkringle containing transmembrane protein 1
1 - 1 of 1
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Figures
Figure Gallery (1 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
kremen1nl10/nl10; zf106Tgstandard conditionsDay 5
kremen1nl10/nl10; zf106Tgstandard conditionsDay 5
1 - 2 of 2
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
nl10
    		Point Mutation
    s356tTgTransgenic Insertion
      zf106TgTransgenic Insertion
        1 - 3 of 3
        Show
        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Fish
        Antibodies
        Orthology
        Engineered Foreign Genes
        Marker Marker Type Name
        EGFPEFGEGFP
        GFPEFGGFP
        1 - 2 of 2
        Show
        Mapping
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        Citation
        Mulvaney, J.F., Thompkins, C., Noda, T., Nishimura, K., Sun, W.W., Lin, S.Y., Coffin, A., Dabdoub, A. (2016) Kremen1 regulates mechanosensory hair cell development in the mammalian cochlea and the zebrafish lateral line. Scientific Reports. 6:31668.