biostudies-literatureYi JFoundation for the National Institutes of HealthNIGMS NIH HHSe1010642https://www.ebi.ac.uk/biostudies/studies/S-EPMC10022797biostudies-literatureUnknown19(3)Bicaudal D2 (BICD2) is responsible for recruiting cytoplasmic dynein to diverse forms of subcellular cargo for their intracellular transport. Mutations in the human BICD2 gene have been found to cause an autosomal dominant form of spinal muscular atrophy (SMA-LED2), and brain developmental defects. Whether and how the latter mutations are related to roles we and others have identified for BICD2 in brain development remains little understood. BICD2 interacts with the nucleoporin RanBP2 to recruit dynein to the nuclear envelope (NE) of Radial Glial Progenitor cells (RGPs) to mediate their well-known but mysterious cell-cycle-regulated interkinetic nuclear migration (INM) behavior, and their subsequent differentiation to form cortical neurons. We more recently found that BICD2 also mediates NPLoS geneticsRole of Nesprin-2 and RanBP2 in BICD2-associated brain developmental disorders.PMC10022797R01 GM144578R01 GM132478R15 GM128119Yi JNoell CRSolmaz SRVallee RBZhao XHelmer PfalseRole of Nesprin-2 and RanBP2 in BICD2-associated brain developmental disorders.Bicaudal D2 (BICD2) is responsible for recruiting cytoplasmic dynein to diverse forms of subcellular cargo for their intracellular transport. Mutations in the human BICD2 gene have been found to cause an autosomal dominant form of spinal muscular atrophy (SMA-LED2), and brain developmental defects. Whether and how the latter mutations are related to roles we and others have identified for BICD2 in brain development remains little understood. BICD2 interacts with the nucleoporin RanBP2 to recruit dynein to the nuclear envelope (NE) of Radial Glial Progenitor cells (RGPs) to mediate their well-known but mysterious cell-cycle-regulated interkinetic nuclear migration (INM) behavior, and their subsequent differentiation to form cortical neurons. We more recently found that BICD2 also mediates N2023-01-01T00:00:00Z2023 Mar2026-04-08T12:56:34.079Z2024-11-11T19:01:53.268ZS-EPMC100227973693059510.1371/journal.pgen.1010642