NDEL1 is a dynein regulator essential for microtubule organization and neuronal migration. Functionally, NDEL1 organizes the cellular microtubule array and anchors microtubules at the centrosome by targeting the severing protein KATNA1 1. NDEL1 positively regulates dynein activity through complex mechanisms: it recruits LIS1 to dynein and promotes dynein-dynactin-adaptor complex formation 2, though it can also sequester LIS1 away from dynein-binding sites under certain conditions 3. NDEL1 facilitates dynein-dependent processes including Golgi integrity maintenance, vesicle transport, and nucleus-centrosome coupling 1. During brain development, NDEL1 is critical for neuronal migration from the ventricular zone to the cortical plate, operating through nucleus-centrosome coupling mechanisms distinct from its paralog NDE1 4. NDEL1 also regulates neurofilament polymerization and neurite outgrowth in partnership with DISC1 5. Disease relevance includes lissencephaly-associated variants disrupting NDEL1-LIS1 binding 4, schizophrenia susceptibility through DISC1 interactions 6, and glioblastoma overexpression correlating with poor survival 7. Clinical significance stems from NDEL1's role as a potential schizophrenia biomarker with unique oligopeptidase activity 5, making it valuable for understanding cortical malformation pathogenesis and psychiatric disease mechanisms.