In view of the multiple possible mechanisms of C9ORF72 mediated toxicity including non‐cell autonomous effects, it will be of interest to examine cocultures for motorneuron toxicity as has been shown for astrocytes 69. Our findings are specific to the process of oligodendrocyte‐lineage cell development and do not address myelination. In this respect, evaluation of mutant OPCs following transplantation into the non‐myelinating Shiverer mouse along with in vitro study of myelination upon coculture with neurons will be important. Finally, future studies may consider the extent to which genetic causes of ALS are heterogeneous with respect to the properties of oligodendrocytes.