There are conflicting reports about whether Prmt1 and arginine methylation increase or reduce cytoplasmic Fus and Fus-positive cytosolic inclusions (28–33), but we demonstrate here that the loss of Prmt1 function leads to increased aggregation of Fus in the cytoplasm, and that Oxr1-mediated reduction in wild-type and ALS mutant Fus aggregation requires Prmt1 function. The gene discussed is OXR1; the disease is amyotrophic lateral sclerosis.