SKOR1 is a transcriptional corepressor that regulates gene expression through sequence-specific DNA binding and interaction with transcription factor complexes 1. It acts as a corepressor of the LBX1 transcription factor and plays a critical role in regulating interneuron cell fate in the dorsal spinal cord 1. SKOR1 inhibits BMP-SMAD-dependent signaling, a pathway essential for dopaminergic neuronal survival and growth 2. SKOR1 dysregulation is strongly associated with restless legs syndrome (RLS), a common sleep-related sensory-motor disorder 134. Multiple GWAS have identified RLS-associated variants in SKOR1 noncoding regions across diverse populations 145. Gene expression studies demonstrate that SKOR1 regulates pathways relevant to RLS pathogenesis, including neurodevelopment and iron metabolism 1. Notably, MEIS1, another RLS-associated gene, positively regulates SKOR1 expression through direct promoter binding 6. In Parkinson's disease models, α-synuclein overexpression upregulates SKOR1, which impairs neurite growth and reduces mitochondrial respiration capacity 2. However, RLS genetic variants do not confer increased Parkinson's disease risk, suggesting these disorders are genetically distinct entities 7. SKOR1 is highly expressed in the central nervous system and localizes to neuronal cell bodies and dendrites 1.