INSYN1 is a scaffolding component of the protein machinery at inhibitory synapses that regulates GABAergic neurotransmission and postsynaptic inhibition. Based on limited published evidence, INSYN1 is a critical component of the dystroglycan complex (DGC) essential for synaptic organization 1. INSYN1 loss disrupts GABAergic synapse composition, alters excitatory/inhibitory balance, reduces GABA receptor distribution, and impairs cognitive behaviors including fear conditioning memory in mice 1. These findings suggest INSYN1 dysfunction may contribute to cognitive impairments associated with dystroglycan complex mutations.