The image shows the morphology of pyramidal neuron filled with biocytin during the recording from mutant cell.This study focuses on understanding the pathophysiological mechanisms of KCNQ-DEE, a severe form of neonatal epilepsy associated with variants in genes of the KCNQ family. KCNQ genes encode subunits of KV7 voltage-gated potassium channels. Each channel is formed by four subunits and particularly heterotetrametric KV7.2/KV7.3 or KV7.5/KV7.3 channels, carrying the so-called M-current, play a crucial role in modulating neuronal excitability in the central nervous system. Most pathogenic variants have been identified in KCNQ2 and were initially linked to benign familial neonatal convulsions (SLFNE) but over the years additional variants have been identified that are associated with KCNQ-DEE.The research utilizes the KV7.2Thr274Met/+ knock-in mouse model, mirroring the human KCNQ2-DEE phenotype. This model exhibits spontaneous seizures and cognitive deficits, and altered neuronal excitability has recently been demonstrated in the cortex. The current study aims to investigate the functional impact of the p.Thr274Met variant on the electrophysiological properties of CA1 hippocampal pyramidal neurons and interneurons in various postnatal time windows.Retigabine, an activator of KV7 channels, was previously withdrawn from the market due to severe side effects. C60, synthesized to overcome these drawbacks, presents improved stability and prolonged half-life. Building on our electrophysiological data we are investigating whether C60 reverts altered neuronal excitability identified in KV7.2Thr274Met/+ mice. This research contributes to a better understanding of the molecular basis of KCNQ-DEE and explores a promising therapeutic avenue for treating drug-resistant seizures.