WDR62 and microcephaly: We investigated WDR62 function using a mouse model and dermal fibroblasts from affected (homozygote) and unaffected (heterozygote) members of a family carrying a novel truncating mutation in WDR62. Our studies demonstrated that disruption of WDR62 impairs proliferation of neocortical progenitors during late neurogenesis underlain by abnormalities in asymmetric centrosome inheritance causing microcephaly in mice, and impairs mitotic cycle progression in patient-derived fibroblasts, which, similar to mouse neocortical progenitors, transiently arrest at prometaphase.