Research ArticleDevelopment

The Rfx4 Transcription Factor Modulates Shh Signaling by Regional Control of Ciliogenesis

Sci. Signal.  03 Nov 2009:
Vol. 2, Issue 95, pp. ra70
DOI: 10.1126/scisignal.2000602

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Regional Regulation of Cilia Formation

Development of the central nervous system (CNS) requires the activation of transcriptional networks in a precise spatial and temporal pattern. Sonic hedgehog (Shh) signaling, which regulates the activity of the Gli family of transcriptional activators and repressors, is important for CNS development. Shh signaling involves a cellular structure called the primary cilium. Ashique et al. show that the transcription factor Rfx4 serves as a regionally specific regulator of the formation of the primary cilia in the developing CNS. Loss of its activity in mice results in aberrant Shh signaling and Gli3 activity, resulting in defective spinal cord and telencephalon development. Furthermore, the activity of Rfx4 may be regulated by phosphorylation, thus allowing Rfx4 to serve as an upstream regulator of Shh signaling and Gli3 activity in response to developmental signals.