Callosal agenesis and pontine hypoplasia has been reported in the ‘tubulinopathies’ due to mutations in the neuron-specific α- and β-tubulin genes (Cushion et al., 2013), however, additional neuroradiological features (including basal ganglia abnormalities, cerebellar hypoplasia, and complex cortical malformations) commonly found in these disorders were not typically seen in EPG5-related Vici syndrome. Here, EPG5 is linked to tubulinopathy.