GARS1 and neuropathy: The finding that the ISR is similarly activated in both Gars and Yars mutant mice suggests that this mechanism may be in play across the tRNA synthetase-associated neuropathies, though this remains to be tested for mutations in HARS, WARS and AARS. Furthermore, though the Gars/CMT2D mouse models accurately recapitulate the human disease, it also remains to be determined whether the same ISR mechanism is active in human motor and sensory neurons.