Both male and female Adgrl2+/− adult mice displayed microcephaly, affecting mainly the telencephalon, although more severe in Adgrl2+/− female mice, with a defect in anteroposterior growth of the vermis, in line with ADGRL2 expression during telencephalic and cerebellar development in human embryos and fœtuses as well as in chicks and mice. Here, ADGRL2 is linked to microcephaly.