CAMKMT (calmodulin-lysine N-methyltransferase) catalyzes trimethylation of lysine-116 in calmodulin, a key signaling protein involved in calcium-dependent cellular processes. The enzyme localizes to multiple cellular compartments including the nucleoplasm, cytoplasm, and nucleolus, where it participates in protein methylation and regulation of opsin-mediated signaling pathways 1. Clinically, CAMKMT is relevant to hypotonia-cystinuria syndrome, a rare neuromuscular disorder. Mutations in CAMKMT cause atypical hypotonia-cystinuria syndrome, distinguished from the classical form by preserved SLC3A1 function, featuring neonatal hypotonia, ptosis, feeding difficulties, and variable neuromuscular and autonomic manifestations 2. Genome-wide association studies have identified CAMKMT variants associated with anxiety disorder risk, particularly in European ancestry populations. The SNP rs1067327 within CAMKMT on chromosome 2 showed genome-wide significant association with anxiety disorder factor scores 3, with replication in independent military cohorts 4. However, a pilot study examining CAMKMT expression in depressed individuals found no significant differences compared to controls 5, suggesting phenotype-specific genetic effects. CAMKMT variants have also been identified in pharmacogenetic studies of dolutegravir exposure 6 and selection signatures in salt-adapted livestock 7, indicating broader biological relevance beyond neuropsychiatric function.