MYO5A encodes a processive actin-based motor protein that hydrolyzes ATP to power movement along actin filaments in large steps approximating the 36-nm pseudo-repeat structure 1. Its primary functions include melanosome transport to the cell periphery through interaction with the adaptor protein melanophilin 2, and regulation of vesicular transport to the plasma membrane. MYO5A mediates autophagosome-lysosome fusion critical for autophagic flux through direct LC3 binding via two LIR motifs in its coiled-coil and globular tail domains 3. Additionally, MYO5A interacts with the endosomal protein RAB5A effector ANKFY1 to facilitate early endosome transport 4 and links to mitochondrial homeostasis 4. MYO5A mutations cause Griscelli syndrome type 1, characterized by immune dysfunction and neurological deterioration 5. Loss of MYO5A expression causes severe neuropathology including alpha-synuclein and tau hyperphosphorylation, dopaminergic neurodegeneration, impaired mitochondrial respiration, and progressive movement disorders 6. A spontaneous Myo5a splice variant in mice causes rapidly progressive ataxia with cerebellar abnormalities and perinatal lethality 4. MYO5A has emerged as a fusion partner in melanocytic tumors, including Spitz neoplasms and melanomas 78. MYO5A also functions in spermiogenesis through BAG5-mediated protein folding during sperm head-tail coupling apparatus assembly 9.