GABRA1 encodes the alpha-1 subunit of gamma-aminobutyric acid type A receptors (GABAARs), heteropentameric ligand-gated chloride channels that mediate primary inhibitory neurotransmission in the brain 1. The alpha-1 subunit forms the GABA binding site at interfaces with beta subunits; GABA activation opens the channel, allowing selective chloride influx that hyperpolarizes neurons and reduces action potential generation 1. Alpha-1-containing GABAARs are predominantly synaptic and exhibit synaptogenic activity, contributing to inhibitory synapse assembly 1. GABRA1 mutations cause developmental and epileptic encephalopathies with variable phenotypes; de novo mutations in GABRA1 have been identified in infantile spasms, Lennox-Gastaut syndrome, and epilepsy of infancy with migrating focal seizures 23. Pathogenic variants predominantly localize to transmembrane domains and extracellular regions, with transmembrane mutations generally producing more severe phenotypes than N-terminal variants 45. All GABRA1 variants identified in large epilepsy cohorts (n>8500) were de novo, indicating dominant inheritance 6. Mutations impair receptor expression and reduce GABA-evoked current amplitudes 1. Additionally, petrosal GABRA1 neurons detect prostaglandins and mediate influenza-induced sickness behavior via an airway-to-brain sensory pathway 7.