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

Laura Anne Lowery, Jamie Rubin, Hazel Sive

Research output: Contribution to journalArticlepeer-review

52 Scopus citations


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
Issue number5
StatePublished - May 2007
Externally publishedYes


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

ASJC Scopus subject areas

  • Developmental Biology


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