KIF5C is a microtubule-associated motor protein essential for neuronal development and function. As a plus-end-directed kinesin motor, it hydrolyzes ATP to generate force for anterograde axonal and dendritic transport of organelles, cargo proteins, and mRNAs 1. KIF5C is particularly critical for transporting the scaffolding protein MAPK8IP3/JIP3, which is required for axon elongation. The protein is highly expressed during early cortical development in both mice and human forebrain 2. KIF5C mutations cause neurodevelopmental disorders through multiple pathogenic mechanisms. Deletions affecting the ATP-binding domain impair ATPase activity and reduce peroxisome transport capacity 3. Loss-of-function leads to abnormal cortical neuronal migration, reduced dendritic branching, and impaired dendritic spine growth 2. KIF5C deficiency also disrupts synaptic transmission by affecting the balance between excitatory and inhibitory neurotransmission 1. Pathogenic KIF5C variants cause infantile-onset epilepsy, intellectual disability, brain atrophy, and cortical malformations, with early clinical presentation correlating with severity 3. The gene has also been identified in congenital cranial dysinnervation disorders 4. Additionally, KIF5C supports neuronal differentiation of mesenchymal stem cells, regulated by the miR-543 pathway 5. These findings establish KIF5C as a critical regulator of cortical development and neuronal function, with therapeutic implications for neurodevelopmental disease management.