WDR6 (WD repeat domain 6) is a WD-repeat protein containing 11 WD-repeat units organized into two clusters separated by a putative transmembrane domain 1. Its primary function involves 2'-O-ribose methylation of tRNA nucleotides at the wobble position 34 of the anticodon loop in collaboration with methyltransferase FTSJ1, where WDR6 is essential for correct substrate tRNA positioning 2. WDR6 also suppresses amino acid starvation-induced autophagy and enhances STK11/LKB1-mediated growth suppression through enzyme-substrate adaptor activity 3. WDR6 exhibits broad disease relevance across multiple psychiatric and neurological conditions. Genome-wide association studies identified WDR6 as a likely causal candidate gene for obsessive-compulsive disorder, with genetic risk associated with excitatory neurons in the hippocampus and dopamine receptor-containing neurons 45. WDR6 expression increases are genetically associated with anorexia nervosa risk, supported by multiple conditional analyses and fine-mapping 6. Additionally, WDR6 was identified as a candidate Parkinson disease gene through integrated GWAS and expression analyses 7. Clinically, WDR6 functions as a prognostic biomarker in lung cancer, where high expression correlates with improved overall survival and immune cell infiltration, particularly in adenocarcinoma 8. WDR6 also acts as a viral restriction factor against vaccinia virus mutants deficient in immune evasion genes 3.