CDKL5 and craniodiaphyseal dysplasia: Our results suggest that CDKL5, through its interaction with the inhibitory scaffolding complex containing gephyrin and CB, regulates membrane levels of synaptic γ2-containing GABAARs. Deranged GABAAR levels are frequently linked to cognitive deficits and epilepsy, and we speculate that our results may help in explaining the seizure phenotype observed in CDD patients.