DNM1 (dynamin 1) is a GTPase that catalyzes vesicle scission through GTP hydrolysis, functioning as a master regulator of multiple endocytic pathways 12. The protein self-assembles into helical polymers around vesicle necks through GTP-dependent oligomerization, constricting membrane invaginations until polymer-induced membrane tubule narrowing triggers fission 34. DNM1 regulates clathrin-mediated endocytosis, synaptic vesicle recycling, and rapid calcium-dependent endocytosis, with particular importance in controlling vesicle size at hippocampal synapses 56. The protein achieves maximum expression in the central nervous system, reflecting its critical neuronal functions 7. DNM1 mutations cause developmental and epileptic encephalopathy (DEE), characterized by severe early-onset seizures and intellectual disability when affecting the GTPase or middle domains 8. Patients display atypical phenotypes including focal epilepsy with milder cognitive impairment when variants occur in alternative domains 8. DNM1 encephalopathy presents with neuroimaging abnormalities including delayed myelination, white matter abnormalities, and cerebral atrophy, likely stemming from GABAergic neuron dysfunction and impaired axonal development 9. Gene therapy using knockdown-replace strategies shows promise in correcting both seizures and synaptic transmission deficits in disease models 10. Exome sequencing confirms DNM1's role in epilepsy diagnosis, with over 50 reported disease cases 119.