In mouse, Drosophila, and human cell models of Charcot-Marie-Tooth (CMT) disease, a genetic disorder that causes peripheral neuropathies, tRNA synthetases carry partially dysfunctional mutations that limit the synthesis of charged tRNAs, leading to translational stress and constitutive activation of GCN2 [48,49,50,51]. The gene discussed is EIF2AK4; the disease is Charcot-Marie-Tooth disease.