ANKS1B encodes AIDA-1, a postsynaptic effector protein that plays critical roles in synaptic function and neurodevelopment 1. The protein regulates NMDA receptor subunit composition by facilitating GluN2B transport from the endoplasmic reticulum to synapses, which is essential for NMDAR-dependent synaptic plasticity 2. AIDA-1 associates with GluN2B and transport machinery including KIF17, and its loss leads to reduced GluN2B-mediated transmission and impaired hippocampal plasticity 2. Beyond neuronal function, ANKS1B also controls endothelial barrier function by regulating permeability independently of angiogenesis 3. Clinically, heterozygous ANKS1B deletions cause ANKS1B neurodevelopmental syndrome (ANDS), characterized by autism spectrum disorder, ADHD, and speech/motor deficits 1. Interestingly, social behavioral deficits in ANDS may arise from oligodendrocyte dysfunction rather than solely neuronal effects, as oligodendrocyte-specific Anks1b loss recapitulates social preference deficits 4. The gene has emerged as a significant locus in GWAS for antipsychotic drug response, cognitive ability, and various neuropsychiatric phenotypes 5. Mouse models demonstrate that Anks1b knockout causes prepulse inhibition deficits, hyperactivity, and altered ketamine sensitivity, supporting its role in glutamatergic signaling 6.