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Related Experiment Video

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In Vivo Modeling of the Morbid Human Genome using Danio rerio
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Zebrafish atoh8 mutants do not recapitulate morpholino phenotypes.

Elsie S Place1, James C Smith1

  • 1Developmental Biology Laboratory, Francis Crick Institute, London, United Kingdom.

Plos One
|February 10, 2017
PubMed
Summary

Atoh8 is a bHLH transcription factor. Zebrafish homozygous for the atoh8sa1465 loss-of-function allele show normal development and survival, indicating Atoh8 is dispensable for zebrafish embryogenesis.

Area of Science:

  • Developmental biology
  • Genetics
  • Molecular biology

Background:

  • Atoh8 is a bHLH transcription factor involved in cell differentiation.
  • Previous studies on Atoh8 loss-of-function in mice and zebrafish yielded conflicting results.
  • The precise role of Atoh8 in vertebrate development remains unclear.

Purpose of the Study:

  • To genetically characterize zebrafish embryos homozygous for the atoh8sa1465 loss-of-function allele.
  • To provide genetic evidence for the developmental role of Atoh8 in zebrafish.
  • To clarify the phenotypic consequences of Atoh8 loss in zebrafish.

Main Methods:

  • Generated zebrafish embryos homozygous for the atoh8sa1465 loss-of-function allele.
  • Assessed embryonic morphology, swimbladder inflation, heart looping, and survival to adulthood.

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  • Evaluated pericardial edema, sensitivity to Fog1 loss, vascular patterning, and primitive hematopoiesis.
  • Main Results:

    • Homozygous atoh8sa1465 zebrafish embryos exhibited normal body morphology, swimbladder inflation, and heart looping.
    • These embryos survived to adulthood without pericardial edema or sensitization to Fog1 loss.
    • Vascular patterning and primitive hematopoiesis were unaffected in atoh8sa1465/sa1465 mutant embryos.

    Conclusions:

    • Atoh8 is dispensable for normal zebrafish development under standard laboratory conditions.
    • The previously described pericardial edema phenotype is not a specific consequence of Atoh8 loss.
    • This study provides genetic evidence clarifying the role of Atoh8 in zebrafish development.