OSBPL1A encodes an oxysterol-binding protein that functions as a lipid transport and regulatory protein with diverse cellular roles. The protein binds multiple lipid species including phosphatidic acid, phosphatidylinositol 3-phosphate, 25-hydroxycholesterol, and cholesterol, facilitating intracellular lipid transport and homeostasis 1. OSBPL1A localizes to late endosomes/lysosomes through interaction with RAB7, where it regulates cholesterol efflux and the reverse cholesterol transport pathway 1. Loss-of-function mutations in OSBPL1A impair ABCA1-mediated cholesterol efflux to lipid-free apoA-I and are associated with low HDL-cholesterol levels, suggesting a critical role in HDL metabolism 1. The gene shows tissue-specific imprinted expression patterns in bovines, with isoform-specific monoallelic expression regulated by promoter methylation 2. Clinically, damaging variants in OSBPL1A are significantly enriched in individuals with hypoalphalipoproteinemia 3, and the gene has been implicated in various pathological conditions including ADHD symptom progression 4, premature ovarian insufficiency 5, and epilepsy pathogenesis through mitochondrial dysfunction pathways 6. These findings establish OSBPL1A as an important regulator of lipid metabolism with potential therapeutic implications.