CHD2 is an ATP-dependent chr15-remodeling factor that regulates gene expression through histone modification and chr15 remodeling 1. As a member of the sucrose nonfermenting protein family, CHD2 binds to gene promoters and mediates histone H3.3 deposition to regulate transcription 2. CHD2 coordinates with transcription factors like FOSL1 to control expression of axon-guidance and synaptic genes 3. In the nervous system, CHD2 serves as an FMRP target involved in neurogenesis and neuronal maturation 2. De novo loss-of-function mutations in CHD2 cause developmental and epileptic encephalopathy 94, characterized by early-onset seizures, developmental delay, and intellectual disability 1. CHD2 mutations account for approximately 1.2% of epileptic encephalopathy cases 4 and have been identified in infantile spasms, Lennox-Gastaut syndrome, and epilepsy with eyelid myoclonia (Jeavons syndrome) 56. Additionally, CHD2 has bidirectional dosage sensitivity, as increased CHD2 expression resulting from CHASERR lncRNA deletion also causes neurodevelopmental disorder with encephalopathy, cortical atrophy, and hypomyelination 7. Beyond epilepsy, CHD2 drives neuron-glioma interactions in pediatric diffuse midline gliomas 3. CHD2 mutations contribute to moderate-to-severe intellectual disability 8, establishing CHD2 as a critical regulator of nervous system development and function.