Autosomal recessive variants in TUBGCP2 encoding the γ-tubulin complex 2 (GCP2) protein were first reported in 5 individuals from 4 families with developmental delay, dysmorphic features, hypotonia, epilepsy, microcephaly, and lissencephaly spectrum changes on brain magnetic resonance imaging (pachygyria, agyria, subcortical band heterotopia), representing defective neuronal migration (Mitani et al., 2019). Here, TUBGCP2 is linked to lissencephaly spectrum disorders.