ArticleCurrent biology : CB2010
Cse1l is a negative regulator of CFTR-dependent fluid secretion.
Article in Current biology : CB, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 36 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
36 citing papers in PubMed, 48 citations in OpenAlex.
- Cystic fibrosis: insights from zebrafish models.Disease models & mechanisms · 2026Review
- The Mechanics of Building Functional Organs.Cold Spring Harbor perspectives in biology · 2025Review
- Polarized transport of membrane and secreted proteins during lumen morphogenesis.Seminars in cell & developmental biology · 2023Review
- Nuclear Factor of Activated T Cells-5 Regulates Notochord Lumenogenesis in Chordate Larval Development.International journal of molecular sciences · 2022Article
- Article
- Patient personalized translational tools in cystic fibrosis to transform data from bench to bed-side and back.American journal of physiology. Gastrointestinal and liver physiology · 2021Article
- What Role Does CFTR Play in Development, Differentiation, Regeneration and Cancer?International journal of molecular sciences · 2020Review
- Towards next generation therapies for cystic fibrosis: Folding, function and pharmacology of CFTR.Journal of cystic fibrosis : official journal of the European Cystic Fibrosis Society · 2020Review
- Osmotic Gradients in Epithelial Acini Increase Mechanical Tension across E-cadherin, Drive Morphogenesis, and Maintain Homeostasis.Current biology : CB · 2020Article
- Article
- Lumen Expansion Facilitates Epiblast-Primitive Endoderm Fate Specification during Mouse Blastocyst Formation.Developmental cell · 2019Article
- CSE1L participates in regulating cell mitosis in human seminoma.Cell proliferation · 2019Article
- Article
- CFTR mutation enhances Dishevelled degradation and results in impairment of Wnt-dependent hematopoiesis.Cell death & disease · 2018Article
- Cell volume changes contribute to epithelial morphogenesis in zebrafish Kupffer's vesicle.eLife · 2018Article
- Quantitative trait loci mapping for feed conversion efficiency in crucian carp (Carassius auratus).Scientific reports · 2017Article
- The expression of water channel proteins during human salivary gland development: a topographic study of aquaporins 1, 3 and 5.Journal of molecular histology · 2017Article
- Claudins in morphogenesis: Forming an epithelial tube.Tissue barriers · 2017Review
- Sheath Cell Invasion and Trans-differentiation Repair Mechanical Damage Caused by Loss of Caveolae in the Zebrafish Notochord.Current biology : CB · 2017Article
- A holey pursuit: lumen formation in the developing kidney.Pediatric nephrology (Berlin, Germany) · 2017Review
Corrections and comments
- Erratum issued
Authors and funding
9 authors at 3 institutions in 1 country.
Funding
Abstract
Transport of chloride through the cystic fibrosis transmembrane conductance regulator (CFTR) channel is a key step in regulating fluid secretion in vertebrates [1, 2]. Loss of CFTR function leads to cystic fibrosis [1, 3, 4], a disease that affects the lungs, pancreas, liver, intestine, and vas deferens. Conversely, uncontrolled activation of the channel leads to increased fluid secretion and plays a major role in several diseases and conditions including cholera [5, 6] and other secretory diarrheas [7] as well as polycystic kidney disease [8-10]. Understanding how CFTR activity is regulated in vivo has been limited by the lack of a genetic model. Here, we used a forward genetic approach in zebrafish to uncover CFTR regulators. We report the identification, isolation, and characterization of a mutation in the zebrafish cse1l gene that leads to the sudden and dramatic expansion of the gut tube. We show that this phenotype results from a rapid accumulation of fluid due to the uncontrolled activation of the CFTR channel. Analyses in zebrafish larvae and mammalian cells indicate that Cse1l is a negative regulator of CFTR-dependent fluid secretion. This work demonstrates the importance of fluid homeostasis in development and establishes the zebrafish as a much-needed model system to study CFTR regulation in vivo.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.