Highlights We performed transcriptome analysis of hiPSC-derived ALS FUS mutant motoneurons Bioinformatics analysis revealed cell adhesion pathways associated to sporadic ALS miR-375 expression is affected by FUS mutation in motoneurons Impairment of miR-375 alters RNA metabolism and increases pro-apoptotic factors Summary The FUS gene has been linked to amyotrophic lateral sclerosis (ALS). FUS is a ubiquitous RNA-binding protein, and the mechanisms...