Search bioRxiv⌕ Search

bioRxiv · 10.64898/2026.08.18.745541

Regulation of Mechanosensor PIEZO Channel Trafficking in C. elegans Germline

Abstract

PIEZO proteins are essential mechanotransductive ion channels, yet mechanisms governing their subcellular localization and membrane trafficking remain elusive. Here, we leverage the C. elegans germline as an in vivo imaging platform to decipher the molecular networks directing PIEZO channel dynamics. High-resolution confocal imaging of endogenously tagged PEZO-1, the C. elegans ortholog, reveals that PEZO-1 compartmentalizes with caveolae protein CAV-1 and the recycling endosome regulator RAB-11. Depletion of rab-11 or the t-SNARE syx-4 severely impairs the translocation of PEZO-1-positive vesicles to the plasma membrane, establishing a reliance on conserved, RAB-11-dependent machinery. Furthermore, we demonstrated that PEZO-1 structural integration is essential for vesicle formation and transport; truncating either the N-terminal transmembrane domains or the C-terminal ion pore induces aberrant vesicle morphology and arrests trafficking. Crucially, PEZO-1 vesicle formation and transport are non-autonomously modulated by reproductive signals requiring male sperm or major sperm protein (MSP) signaling. Finally, introducing conserved disease-associated pathogenic PIEZO mutations markedly suppresses its cytosolic and plasma membrane expression. Collectively, our findings define a fundamental cytological framework regulating PIEZO channel dynamics, shedding light on the molecular etiology of PIEZO-associated channelopathies.

Explore related subjects

Keep this discovery

Explore connections, maps & timelines

BibTeXRIS

Rincon Paz, M., Wang, Y., Shao, E., Monsalve, S., Bai, X.. 2026-08-20. Regulation of Mechanosensor PIEZO Channel Trafficking in C. elegans Germline. https://doi.org/10.64898/2026.08.18.745541

Cite the original work for its findings. Save a collection to share your selection of sources.

KEEP EXPLORING

Related preprints

OPA1 controls mitochondrial dysfunction-driven liver fibrosis in MASLD

Progressive hepatic fibrosis is the principal determinant of morbidity and mortality in metabolic dysfunction-associated steatotic liver disease and steatohepatitis (MASLD/MASH). Mitochondrial dysfunction is a hallmark of MASH, and the release of mitochondrial damage-associated molecular patterns (mito-DAMPs) from injured hepatocytes can promote fibrosis. However, how mitochondrial dynamics and quality control shape the fibrotic response in MASLD/MASH remains unclear. Here, through large-scale genomic analyses of mitochondrial genes governing mitophagy, fusion and fission in human MASLD, with a power-equivalent sample size of approximately 700,000 individuals, we identify a strong association between hepatic fibrosis and the mitochondrial fusion factor dynamin-like GTPase optic atrophy 1 (OPA1). OPA1 transcripts and protein abundance in the liver epithelium were progressively dysregulated with advancing fibrosis. In mice, hepatocyte-specific OPA1 loss alone was sufficient to induce hepatic stellate cell activation and fibrosis in zone 3, promoted the release of mito-DAMPs into the circulation and exacerbated fibrosis in experimental MASH. These findings identify OPA1 as a central regulator of the hepatic fibrotic response and connect defective mitochondrial homeostasis to mito-DAMP release, hepatic stellate cell activation and fibrosis in MASLD.

genetics↗

Mechanism-selective deep mutational scanning distinguishes ERCC2 disease phenotypes

Pathogenic ERCC2 variants cause xeroderma pigmentosum (XP), trichothiodystrophy (TTD) or both, yet variant effect scores are usually interpreted only as measures of pathogenicity rather than of which disease mechanism is disrupted. XPD, the ERCC2-encoded TFIIH subunit, functions in both nucleotide excision repair and transcription. Using yeast complementation deep mutational scanning, we measured the effects of nearly all XPD amino acid substitutions. The assay was mechanism-selective: it preferentially reported transcription-associated function, with pronounced intolerance at the p44 interface, whereas many substitutions affecting DNA binding and helicase activity retained near-wild-type fitness. Accordingly, TTD variants had much lower fitness than XP variants. Computational predictors discriminated pathogenic from benign variants similarly across phenotypes, but the DMS distinguished XP from TTD variants better than all 73 predictors tested. Phenotype-specific ACMG/AMP calibration provided evidence in both directions for TTD but mainly pathogenic evidence for XP. Thus, the selectivity of functional assays, often viewed as a limitation, can reveal disease mechanisms and support phenotype-aware variant interpretation.

genetics↗

Temporal control of mitochondrial mutagenesis reveals the fate of mtDNA mutations with age

Mutations in the mitochondrial genome (mtDNA) play a critical role in the aging process and a wide variety of age-related diseases. However, it remains unclear when the mutations that drive physiological decline arise. To answer this question, we generated a new mouse model in which mitochondrial mutagenesis can be confined to a defined window of time. Surprisingly, we found that mutations that arise during the first two months of life are sufficient to drive a wide variety of age-related pathologies, and that the severity of this pathology is broadly regulated by distinct, tissue-specific selective pressures that control the fate of mtDNA mutations with age. Further, we found that selection against deleterious variants can be modulated by manipulation of mitochondrial fusion in vitro and in vivo. These observations raise the possibility that in some tissues, the pace of aging is pre-determined by events that occur early in life and that interventions targeting mitochondrial fusion may be able to slow down or reverse the expansion of these pathogenic variants. These results carry far-reaching implications for strategies aimed at preventing or delaying age-related decline.

genetics↗