In a previous work, we demonstrated that the three established CMT-causing TyrRS mutants (TyrRS-E196K, TyrRS-G41R, and TyrRS-Δ153-156VKQV) induce a conformational opening and expose a consensus area in the catalytic domain of the enzyme17 (Fig. 1a, b). This evidence concerns the gene YARS1 and Charcot-Marie-Tooth disease.