Oxidation resistance 1 (OXR1) has been recently shown to protect neurons from oxidative stress, and it is upregulated in the spinal cord of patients with amyotrophic lateral sclerosis (ALS). To assess the potential neuroprotective properties of OXR1 in ALS, Liu and colleagues crossed SOD1G93A ALS-model mice with mice that overexpress OXR1 in neurons. The offspring had longer survival, attenuated motor deficits, and delayed pathological changes compared with typical SOD1G93A mice, suggesting that OXR1 is a therapeutic target for ALS.