Strikingly, partial deletion of CEP170's centrosomal targeting and microtubule-binding domains led to severe migration deficits in the developing cortex.<h4>Conclusion</h4>Our findings identify CEP170 as a critical regulator of neural progenitor proliferation, neuronal migration, and cortical architecture via centrosome-microtubule interactions, providing new insights into centrosome-linked neurodevelopmental disorders. Here, CEP170 is linked to neurodevelopmental disorder.