Using BMAA to induce ALS-like symptoms, we screened three neuroprotective compounds—CNR-401, Edaravone, and Cannflavin A—for their therapeutic impact. CNR-401 stood out, not only reversing motor deficits but also driving robust molecular changes in key ALS-related pathways like neuroinflammation, ECM remodeling, and PI3K-Akt signaling. Our work underscores the power of integrative RNA-seq approaches (with help from Canurta’s DEG Pipeline Assistant) to link phenotype and gene expression—moving us closer to viable ALS interventions.