TARDBP and amyotrophic lateral sclerosis: Therefore, in this work, we provide the first molecular characterization of whole third instar TBPHΔ23/Δ23 larvae, through which we not only validated its relevance as a TDP-43/TBPH-mediated ALS model even at the transcriptomic level, but we also uncovered novel and hidden pathological mechanisms underlying the disease, thereby opening new pathways for drug discovery.