ArticleBiology open2015
The zebrafish Kupffer's vesicle as a model system for the molecular mechanisms by which the lack of Polycystin-2 leads to stimulation of CFTR.
Article in Biology open, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
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Who cites it
17 citing papers in PubMed, 26 citations in OpenAlex.
- Cystic fibrosis: insights from zebrafish models.Disease models & mechanisms · 2026Review
- Fluid extraction from the left-right organizer uncovers mechanical properties needed for symmetry breaking.eLife · 2023Article
- Zebrafish: A Promising Real-Time Model System for Nanotechnology-Mediated Neurospecific Drug Delivery.Nanoscale research letters · 2021Review
- Zebrafish Model as a Screen to Prevent Cyst Inflation in Autosomal Dominant Polycystic Kidney Disease.International journal of molecular sciences · 2021Article
- Pkd2 Affects Cilia Length and Impacts LR Flow Dynamics andFrontiers in cell and developmental biology · 2021Article
- What Role Does CFTR Play in Development, Differentiation, Regeneration and Cancer?International journal of molecular sciences · 2020Review
- A high throughput zebrafish chemical screen reveals ALK5 and non-canonical androgen signalling as modulators of the pkd2Scientific reports · 2020Article
- Aplnra/b Sequentially Regulate Organ Left-Right Patterning via Distinct Mechanisms.International journal of biological sciences · 2019Article
- Tissue Flow Induces Cell Shape Changes During Organogenesis.Biophysical journal · 2018Article
- CFTR is required for the migration of primordial germ cells during zebrafish early embryogenesis.Reproduction (Cambridge, England) · 2018Article
- Vertebrate myosin 1d regulates left-right organizer morphogenesis and laterality.Nature communications · 2018Article
- Cell volume changes contribute to epithelial morphogenesis in zebrafish Kupffer's vesicle.eLife · 2018Article
- Article
- Spatial Allocation and Specification of Cardiomyocytes during Zebrafish Embryogenesis.Korean circulation journal · 2017Review
- Dynamics of cilia length in left-right development.Royal Society open science · 2017Article
- Identification and Expression Analysis of the Complete Family of ZebrafishFrontiers in cell and developmental biology · 2017Article
- Claudin5a is required for proper inflation of Kupffer's vesicle lumen and organ laterality.PloS one · 2017Article
Corrections and comments
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
In autosomal dominant polycystic kidney disease (ADPKD), cyst inflation and continuous enlargement are associated with marked transepithelial ion and fluid secretion into the cyst lumen via cystic fibrosis transmembrane conductance regulator (CFTR). Indeed, the inhibition or degradation of CFTR prevents the fluid accumulation within cysts. The in vivo mechanisms by which the lack of Polycystin-2 leads to CFTR stimulation are an outstanding challenge in ADPKD research and may bring important biomarkers for the disease. However, hampering their study, the available ADPKD in vitro cellular models lack the three-dimensional architecture of renal cysts and the ADPKD mouse models offer limited access for live-imaging experiments in embryonic kidneys. Here, we tested the zebrafish Kupffer's vesicle (KV) as an alternative model-organ. KV is a fluid-filled vesicular organ, lined by epithelial cells that express both CFTR and Polycystin-2 endogenously, being each of them easily knocked-down. Our data on the intracellular distribution of Polycystin-2 support its involvement in the KV fluid-flow induced Ca(2+)-signalling. Mirroring kidney cysts, the KV lumen inflation is dependent on CFTR activity and, as we clearly show, the knockdown of Polycystin-2 results in larger KV lumens through overstimulation of CFTR. In conclusion, we propose the zebrafish KV as a model organ to study the renal cyst inflation. Favouring its use, KV volume can be easily determined by in vivo imaging offering a live readout for screening compounds and genes that may prevent cyst enlargement through CFTR inhibition.
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