CEP85L encodes a centrosomal protein that plays an essential role in neuronal migration during brain development. The protein localizes to the pericentriolar material of the maternal centriole, where it forms a complex with key migration regulators including CDK5, LIS1, NDE1, KIF2A, and DYNC1H1 1. CEP85L functions by controlling CDK5 localization and activation, which is critical for proper centrosome organization and microtubule cytoskeleton dynamics during neuronal migration 1. Loss of CEP85L disrupts these processes, leading to defective neuronal migration with particularly severe effects in the posterior neocortex 1. Pathogenic variants in CEP85L cause lissencephaly type 10, a neurodevelopmental disorder characterized by posterior predominant lissencephaly with abnormal cortical thickness and absent cerebral convolutions 23. Clinical manifestations include global developmental delay (71% of cases), intellectual disability (74% of cases), and seizures (90% of cases), with brain imaging revealing predominantly posterior lissencephaly often accompanied by subcortical band heterotopia 4. Most pathogenic variants cluster in a highly conserved N-terminal region (amino acids 1-103) and follow autosomal dominant inheritance patterns 42. CEP85L represents an important genetic cause of posterior predominant lissencephaly, highlighting the critical role of centrosomal proteins in cortical development.