Figure 1: FEZ1 is critical for neurodevelopment. In CNS and PNS neurons, FEZ1 participates in both axo-dendritic development as well as synapse formation and organization. (1) FEZ1 acts downstream effector of guidance cue signaling pathways to regulate axo-dendritic development and migration. FEZ1 also interacts with DISC1 or ZNF804A to modulate dendrite length, branching and short spine density. (2) FEZ1/Kinesin-1 complexes transport synaptic components essential for synapse formation and organization. (3) Likewise, FEZ1 role’s in transporting synaptic components during motor neuron development is essential for proper formation of neuromuscular junctions (NMJ). Loss of FEZ1 in these processes eventually leads to neuronal network perturbations and potentially contributes to neurodevelopmental disorders. CNS: Central nervous system; CRMP1: collapsin response mediator protein 1; DCC: deleted in colorectal cancer; DISC1: disrupted in schizophrenia 1; FEZ1: fasciculation and elongation zeta 1; NMJ: neuromuscular junction; NRP1: neuropilin 1; PNS: peripheral nervous system; PTV: piccolo transport vesicle; STX1A: syntaxin-1A; ZNF804A: zinc finger protein 804A. Image was created using Biorender graphic software (https://biorender.com/).