FILIP1 (filamin A interacting protein 1) is a structural protein that regulates cortical neuronal migration and muscle function through interactions with filamin proteins. Mechanistically, FILIP1 controls neocortical cell migration initiation from the ventricular zone via a filamin-A/F-actin axis 1 and mediates filamin-C (FLNc) degradation through direct binding 2. Dual-site phosphorylation of FLNc by Akt and PKCα reduces FILIP1 binding, protecting FLNc from degradation and enabling its signaling function in muscle 2. Clinically, bi-allelic FILIP1 variants cause autosomal recessive disorders with severe manifestations. Patients exhibit congenital myopathy with myofibrillar disintegration, brain malformations, neurodevelopmental delay, and dysmorphic features 1. Homozygous truncating variants cause arthrogryposis multiplex congenita with microcephaly, congenital joint contractures, scoliosis, and facial dysmorphism 3. FILIP1 was identified as a candidate gene associated with intellectual disability in consanguineous Middle Eastern families 4. Additionally, FILIP1 variants show suggestive associations with lipid metabolism and atherosclerosis risk, implicating brain-adipose-liver communication pathways 56, and genome-wide association studies identified FILIP1 loci near osteoarthritis susceptibility regions 7.