C9orf72 KO mice, C9ORF72 patients iPSC-derived, and C9ORF72± iMNs share similar gene expression changes to the postmortem tissue and blood from C9-FTD/ALS patients, in particular regarding the immune and endolysosomal pathways (O’Rourke et al., 2016; Shi et al., 2018; McCauley et al., 2020). Here, C9orf72 is linked to amyotrophic lateral sclerosis.