CAMSAP1 is a key microtubule-organizing protein that specifically recognizes and stabilizes the minus-ends of non-centrosomal microtubules 1. It functions as a minus-end tracking protein (-TIP) that binds at growing microtubule minus-end tips and protects them from depolymerization without significantly affecting polymerization rates 23. Through spectrin interaction, CAMSAP1 regulates neurite outgrowth and is critical for neuronal migration and brain development 45. CAMSAP1 plays essential roles in spermiogenesis by orchestrating manchette microtubule dynamics; its loss disrupts sperm development and causes infertility phenotypes 6. Bi-allelic loss-of-function variants in CAMSAP1 cause a clinically recognizable neuronal migration disorder characterized by severe microcephaly, lissencephaly with posterior gradient, corpus callosum hypoplasia, neurodevelopmental delay, and seizures 5. CAMSAP1 is associated with cortical dysplasia and developmental malformations 7. The gene is also implicated in intellectual disability in consanguineous populations 8 and represents a hub gene in brain aging networks 9. CAMSAP1 expression regulates microtubule formation in cancer contexts, with implications for tumor biology 10.