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CEP170 as a novel molecular link between centrosomal function and cerebral cortical development.

Yu-Ching Liao1, Meng-Han Tsai2,3,4, Nian-Hsin Chao1

  • 1Institute of Brain Science, College of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan.

Journal of Biomedical Science
|March 27, 2026
PubMed
Summary

CEP170 is crucial for brain development, regulating neural progenitor proliferation and neuronal migration. Its dysfunction impacts cortical architecture and is linked to neurodevelopmental disorders.

Keywords:
CCDC120CEP170CentrosomeLissencephalyMicrocephalyMicrotubule organizationNeural progenitorNeuronal migration

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Area of Science:

  • Neuroscience
  • Cell Biology
  • Developmental Biology

Background:

  • The centrosome is vital for brain development, controlling microtubule dynamics and neuronal migration.
  • CEP170, a centriole-associated protein, is implicated in centrosome function but its role in corticogenesis is unclear.

Purpose of the Study:

  • To investigate the role of CEP170 in mammalian cortical development.
  • To elucidate the molecular mechanisms underlying CEP170 function in neurogenesis.

Main Methods:

  • Analyzed CEP170 expression in mouse and human cortex using transcriptomic and proteomic techniques.
  • Assessed loss-of-function phenotypes via in utero electroporation and CRISPR/Cas9 gene editing in mice.
  • Investigated cell proliferation, microtubule dynamics, and protein interactions.

Main Results:

  • CEP170 is expressed in neural progenitors and neurons during cortical development.
  • Cep170 knockdown caused neuronal migration deficits, altered laminar fate, and impaired progenitor proliferation.
  • CEP170 C-terminal truncations disrupted microtubule organization and localization, leading to migration defects.

Conclusions:

  • CEP170 is essential for neural progenitor proliferation and neuronal migration in the developing cortex.
  • CEP170 regulates cortical architecture through centrosome-microtubule interactions.
  • Findings offer insights into centrosome-related neurodevelopmental disorders.