is a versatile platform for small-molecule screening across many disease models. Here, we utilized a model carrying a clinically relevant mutation in Polr1D to test the antioxidant N-acetyl- -cysteine (NAC) for therapeutic potential. Treating heterozygous mothers with NAC partially suppressed the developmental defects of their homozygous mutant larvae. These findings are consistent with antioxidant rescue effects observed in zebrafish and mouse models of Treacher Collins Syndrome (TCS). Our results demonstrate the value of a mutant model for identifying chemical suppressors and accelerating the discovery of promising therapeutics in disorders like TCS.