# Maternal antioxidant treatment partially rescues developmental defects in a Drosophila Polr1D mutant model

## TL;DR

Treating pregnant fruit flies with an antioxidant partially reduced developmental issues in their mutant offspring, suggesting potential therapeutic approaches for similar human disorders.

## Contribution

The study demonstrates that a Drosophila Polr1D mutant model can identify antioxidant-based chemical suppressors for developmental disorders.

## Key findings

- Maternal treatment with NAC partially rescued developmental defects in Polr1D mutant larvae.
- The results align with antioxidant rescue effects observed in zebrafish and mouse models of Treacher Collins Syndrome.
- The Drosophila Polr1D mutant model is effective for identifying chemical suppressors for developmental disorders.

## Abstract

Drosophila melanogaster
is a versatile
in vivo
platform for small-molecule screening across many disease models. Here, we utilized a
Drosophila
model carrying a clinically relevant mutation in Polr1D to test the antioxidant N-acetyl-
L
-cysteine (NAC) for therapeutic potential. Treating heterozygous
Polr1D
mothers with NAC partially suppressed the developmental defects of their homozygous
Polr1D
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
Polr1D
mutant
Drosophila
model for identifying chemical suppressors and accelerating the discovery of promising therapeutics in disorders like TCS.