Whitesnake/sfpq is required for cell survival and neuronal development in the zebrafish

Laura Anne Lowery, Jamie Rubin, Hazel Sive

Research output: Contribution to journalArticle

41 Citations (Scopus)

Abstract

Organogenesis involves both the development of specific cell types and their organization into a functional three-dimensional structure. We are using the zebrafish to assess the genetic basis for brain organogenesis. We show that the whitesnake mutant corresponds to the sfpq (splicing factor, proline/glutamine rich) gene, encoding the PSF protein (polypyrimidine tract-binding protein-associated splicing factor). In vitro studies have shown that PSF is important for RNA splicing and transcription and is a candidate brain-specific splicing factor, however, the in vivo function of this gene is unclear, sfpq is expressed throughout development and in the adult zebrafish, with strong expression in the developing brain, particularly in regions enriched for neuronal progenitors. In the whitesnake mutant, a brain phenotype is visible by 28 hr after fertilization, when it becomes apparent that the midbrain and hindbrain are abnormally shaped. Neural crest, heart, and muscle development or function is also abnormal, sfpq function appears to be required in two distinct phases during development. First, loss of sfpq gene function leads to increased cell death throughout the early embryo, suggesting that cell survival requires functional PSF protein. Second, sfpq function is required for differentiation, but not for determination, of specific classes of brain neurons. These data indicate that, in vertebrates, sfpq plays a key role in neuronal development and is essential for normal brain development.

Original languageEnglish (US)
Pages (from-to)1347-1357
Number of pages11
JournalDevelopmental Dynamics
Volume236
Issue number5
DOIs
StatePublished - May 1 2007
Externally publishedYes

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Zebrafish
Cell Survival
Brain
Organogenesis
Genes
RNA Splicing
Rhombencephalon
Muscle Development
Neural Crest
Mesencephalon
Glutamine
Fertilization
Proline
Vertebrates
Myocardium
Proteins
Cell Death
Embryonic Structures
Phenotype
Neurons

Keywords

  • Brain development
  • Cell death
  • Neuronal determination
  • Neuronal differentiation
  • PSF
  • Splicing factor proline glutamine rich
  • Whitesnake
  • Zebrafish

ASJC Scopus subject areas

  • Developmental Biology

Cite this

Whitesnake/sfpq is required for cell survival and neuronal development in the zebrafish. / Lowery, Laura Anne; Rubin, Jamie; Sive, Hazel.

In: Developmental Dynamics, Vol. 236, No. 5, 01.05.2007, p. 1347-1357.

Research output: Contribution to journalArticle

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