MYO1E encodes an unconventional myosin motor protein with ATPase activity that serves as an actin-based motor molecule 12. The protein binds to anionic phospholipid-containing membranes via its tail domain and plays a central role in clathrin-mediated endocytosis by connecting clathrin-coated vesicles to the actin cytoskeleton 2. MYO1E is highly enriched in podocytes, where it regulates actin cytoskeleton dynamics and cell adhesion critical for maintaining the glomerular filtration barrier 34. Loss-of-function mutations in MYO1E cause steroid-resistant nephrotic syndrome (SRNS), characterized by podocyte dysfunction, disrupted glomerular basement membrane morphology, and proteinuria 53. MYO1E impairment leads to actin reorganization, impaired cell proliferation, migration, endocytosis, and adhesion 4. Pathogenic variants typically localize to the motor and neck domains, with mutations like T119I disrupting subcellular localization and D388H reducing ATPase activity and motor function 2. Beyond renal disease, MYO1E overexpression in lung adenocarcinoma associates with increased mortality risk and altered DNA methylation patterns, suggesting broader oncologic relevance 6.