• MO-mediated flna KD in zebrafish embryos (Adams et al., 2012): Meckel–Gruber syndrome-like phenotype (ciliopathy), with pronephric cysts, hydrocephalus and notochord abnormalities• FAT4 and DCHS deficient human iPSCs and COs (Klaus et al., 2019): patients-derived and isogenic knock-out lines. Altered neuronal morphology and migration abilities, resulting in neurons accumulating in the VZ compartment. Here, FAT4 is linked to ciliopathy.