Highlights • MCPH1 expression is prominent in RGCs during the early stages of neocortical development • Early loss of aRGCs due to Mcph1 mutation induces microcephaly in the mouse neocortex • Mcph1 influences cell proliferation and survival via crosstalk with metabolic pathways • Mcph1 stimulates mitochondrial activity via glutaminolysis and interaction with VDAC1 Summary A distinctive feature of neocortical development is the highly coordinated production of different progenitor cell...