A recent report of 4-AP treatment in patients with KCNA2-encephalopathy has interesting mechanistic implications; 4-AP reduced seizures in patients with gain-of-function variants of the Kv1.2 subunit and antagonized the electrophysiological defects in vitro in transfected neurons that expressed variant Kv1.2 channels [37]. This evidence concerns the gene KCNA2 and Encephalopathy.