GNAO1 encodes the α‐subunit of a heterotrimeric guanine nucleotide‐binding protein (Gαo), which is widely expressed in the central nervous system, playing an important role in signal transduction through AMPc metabolism in the striatum.2, 5, 6 As the number of reports increased, it became evident that GNAO1‐related encephalopathies encompass a continuous spectrum of neurological syndromes featuring variable association of movement disorders, psychomotor delay, intellectual disability (ID), and different types of epilepsy.2, 7GNAO1‐related movement disorder usually starts in infancy. This evidence concerns the gene GNAO1 and epilepsy.