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Related Experiment Video

Updated: May 23, 2025

Quantitative PCR-based Assay to Measure Sonic Hedgehog Signaling in Cellular Model of Ciliogenesis
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Multicilia dynamically transduce Sonic Hedgehog signaling to regulate choroid plexus functions.

Suifang Mao1, Rui Song1, Shibo Jin1

  • 1Division of Cellular and Developmental Biology, MCB Department, University of California, Berkeley, Berkeley, CA 94705, USA.

Cell Reports
|March 9, 2025
PubMed
Summary

Defective choroid plexus cilia cause neonatal hydrocephalus by altering Aqp1 and Atp1a2 expression. Developmental changes in these sensory cilia regulate Sonic Hedgehog (Shh) signaling to control cerebrospinal fluid production.

Keywords:
Aqp1Atp1a2CP: Cell biologyCSFFoxJ1GαiShhSmocAMPcerebrospinal fluidchoroid plexus cilia

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Area of Science:

  • Neuroscience
  • Cell Biology
  • Developmental Biology

Background:

  • The choroid plexus epithelium, featuring multiciliated cells, is crucial for cerebrospinal fluid (CSF) production.
  • Cilia dysfunction is linked to various neurological disorders, including hydrocephalus.

Purpose of the Study:

  • To investigate the role of choroid plexus multicilia in regulating CSF production and their connection to hydrocephalus.
  • To elucidate the signaling pathways involved in choroid plexus ciliary function and development.

Main Methods:

  • Analysis of choroid plexus ciliogenesis and intraflagellar transport in a neonatal hydrocephalus model.
  • Examination of Aqp1 and Atp1a2 expression in relation to ciliary function.
  • Investigation of Sonic Hedgehog (Shh) signaling pathways, including non-canonical pathways involving Smoothened (Smo)/Gαi/cyclic AMP (cAMP).

Main Results:

  • Defective choroid plexus ciliogenesis or intraflagellar transport leads to neonatal hydrocephalus.
  • This condition is associated with increased expression of the water channel Aqp1 and ion transporter Atp1a2.
  • Choroid plexus multicilia function as sensory cilia, transducing Shh signaling, with non-canonical Shh repressing Aqp1 and Atp1a2 via the Smo/Gαi/cAMP pathway.
  • Developmental shortening of choroid plexus cilia reduces Shh signaling, derepresses Aqp1 and Atp1a2, and increases CSF production.

Conclusions:

  • Choroid plexus multicilia play a critical sensory role in regulating CSF production through Shh signaling.
  • The unique developmental dynamics of these cilia, specifically their decreasing length, actively dampen Shh signaling to promote CSF production.
  • Understanding these mechanisms offers insights into hydrocephalus pathogenesis and potential therapeutic targets.