# RP gene haploinsufficiency promotes extra sensory organ formation via a threshold effect

**Authors:** Haiwei Pi, Kuan-Han Chen, Hsin Tu, Chung-Wei Hsu

PMC · DOI: 10.1080/19336934.2025.2606496 · 2026-01-06

## TL;DR

Reduced levels of ribosomal protein genes in fruit flies cause extra sensory organs to form when a certain threshold is crossed.

## Contribution

The study reveals a threshold effect in RP gene haploinsufficiency that leads to ectopic sensory organ formation.

## Key findings

- Strongly affected Minute mutants show a threshold-dependent increase in bristle and CS formation.
- Xrp1–Irbp18 transcriptional dimer is upregulated in sensory organ-promoting Minutes.
- Xrp1 mutation suppresses ectopic CS formation, indicating its regulatory role.

## Abstract

Ribosomal protein (RP) gene haploinsufficiency is a conserved form of ribosome dysfunction across species and underlies a class of disorders known as ribosomopathies. In Drosophila, RP gene haploinsufficiency manifests as the Minute phenotype, characterized by thinner and shorter mechanosensory bristles. The development of both bristles and proprioceptive campaniform sensilla (CS) is initiated by the bHLH proneural proteins Achaete (Ac) and Scute (Sc). By analysing genetic interactions between ac sc mutants and Minute mutants of varying severity, we identified a novel bristle-promoting effect that occurs only in the strongly affected Minutes in which the average bristle length is shorter than a threshold. This threshold-dependent effect also promotes ectopic CS formation in the strong Minutes. Transcriptomic analyses comparing the sensory organ – promoting and non-promoting Minutes revealed significant differences in stress-response pathways, including differentially elevated expression of the Xrp1–Irbp18 transcriptional dimer. Notably, mutation of Xrp1 suppresses the ectopic CS phenotype, indicating a positive regulatory role. These findings reveal a previously unrecognized threshold effect in RP gene haploinsufficiency, in which excessive Xrp1 activity promotes supernumerary sensory organ formation, suggesting a compensatory mechanism that modulates neurogenesis under severe ribosomal stress.

## Linked entities

- **Genes:** BLOC1S3 (biogenesis of lysosomal organelles complex 1 subunit 3) [NCBI Gene 388552], ASAH1 (N-acylsphingosine amidohydrolase 1) [NCBI Gene 427], ERMAP (erythroblast membrane associated protein (Scianna blood group)) [NCBI Gene 114625], Xrp1 (Xrp1) [NCBI Gene 42267], Irbp18 (Inverted repeat binding protein 18 kDa) [NCBI Gene 39243]
- **Proteins:** achaete (achaete-scute complex protein T5), sc (scute), Xrp1 (Xrp1), Irbp18 (Inverted repeat binding protein 18 kDa)
- **Species:** Drosophila (taxon 7215)

## Full-text entities

- **Genes:** Xrp1 (Xrp1) [NCBI Gene 42267] {aka 2515, CG17836, DM28, Dmel\CG17836, Xrp, anon-EST:Liang-2.44}, sc (scute) [NCBI Gene 30982] {aka AS-C T4, AS-C T4sc, CG3827, DROACS2, Dmel\CG3827, EG:198A6.1}, ac (achaete) [NCBI Gene 30981] {aka 990 E5 F1, AS-C, AS-C T5, AS-C T5ac, ASC, Achaete}, RpS17 (Ribosomal protein S17) [NCBI Gene 39088] {aka BcDNA:RE44119, CG3922, D9, Dmel\CG3922, M, M(3)67}
- **Diseases:** ribosome dysfunction (MESH:D006331)
- **Species:** Drosophila melanogaster (fruit fly, species) [taxon 7227]

## Figures

8 figures with captions in the complete paper: https://tomesphere.com/paper/PMC12778873/full.md

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Source: https://tomesphere.com/paper/PMC12778873