Cilia in Nervous System Development, Function, and Disease

Cilia are evolutionarily conserved, microtubule‐based organelles that function as sensory and motility hubs, orchestrating a variety of physiological processes within the mammalian nervous system. In the context of neural development and homeostasis, cilia act as “cellular antennae,” integrating extracellular signals to regulate neuronal migration, axonal guidance, and synaptic plasticity. Despite their ubiquity, the exact mechanisms by which specific ciliary signaling pathways coordinate complex neurodevelopmental events remain incompletely understood. Furthermore, little is known about how spatiotemporal disruptions in ciliary assembly or signaling contribute to diverse clinical phenotypes observed in neurological disorders. This review systematically describes important cilia‐dependent signaling cascades, such as Hedgehog and Wnt, and evaluates their multifaceted roles in maintaining normal neural architecture and function. We further synthesize recent breakthroughs linking ciliary dysfunction to a broad spectrum of ciliopathies, emphasizing the molecular basis of associated cognitive and motor impairments. By identifying limitations in our current understanding and proposing strategic directions for future studies, this review provides a comprehensive framework to help both basic researchers and clinicians decipher ciliary biology and move toward clinical translation. This review also highlights the therapeutic potential of targeting ciliary pathways in severe neurological conditions.