In the developing cerebellum, Sonic hedgehog (SHH) signaling is required for expansion of cerebellar granule neural progenitors, proposed to be cells-of-origin for the SHH-driven pediatric brain tumor medulloblastoma. In this issue of Developmental Cell, Chang et al. (2019) show that the transcription factor Atoh1/MATH1 regulates primary cilium formation, enabling SHH signaling.
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