Evidence map›Paper›PMID 20933420›Full record

ArticleCurrent biology : CB2010

Cse1l is a negative regulator of CFTR-dependent fluid secretion.

Michel Bagnat, Adam Navis, Sara Herbstreith, Koroboshka Brand-Arzamendi, Silvia Curado, Sherif Gabriel, Keith Mostov, Jan Huisken, Didier Y R Stainier

Erratum issuedOpen access · bronzeAbstract read
In one paragraph

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.

0numbers the graph read from it
0cells of the map it votes in
36citing papers in PubMed
2.6field-weighted citation impact, top 10% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

36 citing papers in PubMed, 48 citations in OpenAlex.

  1. Cystic fibrosis: insights from zebrafish models.Disease models & mechanisms · 2026
    Review
  2. The Mechanics of Building Functional Organs.Cold Spring Harbor perspectives in biology · 2025
    Review
  3. Review
  4. Article
  5. Article
  6. 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 · 2021
    Article
  7. Review
  8. 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 · 2020
    Review
  9. Article
  10. Article
  11. Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Article
  18. Review
  19. Article
  20. A holey pursuit: lumen formation in the developing kidney.Pediatric nephrology (Berlin, Germany) · 2017
    Review
4 · The record

Corrections and comments

  • Erratum issued
5 · Who and what money

Authors and funding

9 authors at 3 institutions in 1 country.

Michel BagnatDepartment of Cell Biology, Duke University School of Medicine, Durham, NC 27710, USA. m.bagnat@cellbio.duke.edu
Adam Navis
Sara Herbstreith
Koroboshka Brand-Arzamendi
Silvia Curado
Sherif Gabriel
Keith Mostov
Jan Huisken
Didier Y R Stainier
University of California, San Francisco · USDuke University · USUniversity of North Carolina at Chapel Hill · US

Funding

Vector CoreP30DK065988 · NIDDK · UNIV OF NORTH CAROLINA CHAPEL HILL · PI Richard Charles Boucher, Martina Gentzsch · 2004 to 2026
$26.5M
Sulfs and Injury Repair in Mucosal EpitheliaP01AI053194 · NIAID · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI MOSTOV, KEITH E · 2002 to 2012
$13.4M
MOLECULAR GENETICS OF LIVER DEVELOPMENTR01DK060322 · NIDDK · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI STAINIER, DIDIER Y · 2001 to 2012
$3.8M
Recovery from acute kidney injuryR01DK074398 · NIDDK · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI MOSTOV, KEITH E · 2007 to 2015
$3.0M
Discovering New Regulators of CFTR and Fluid Secretion in ZebrafishDP2OD006486 · OD · DUKE UNIVERSITY · PI BAGNAT, MICHEL · 2009 to 2009
$2.3M
Control of Orientation of Epithelial PolarityR01DK067153 · NIDDK · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI MOSTOV, KEITH E · 2004 to 2008
$1.7M
MOLECULAR GENETICS OF PANCREAS DEVELOPMENTR01DK075032 · NIDDK · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI STAINIER, DIDIER Y · 2007 to 2011
$1.5M
Formation of bile ducts in three dimensional cultureR01DK083330 · NIDDK · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI MOSTOV, KEITH E · 2010 to 2012
$1.0M
NIAID NIH HHS P01 AI053194NIAID NIH HHS P01AI53194NIDDK NIH HHS DK074398NIDDK NIH HHS DK075032NIDDK NIH HHS DK083330NIDDK NIH HHS DK60322NIDDK NIH HHS P30 DK065988NIDDK NIH HHS R01 DK060322NIDDK NIH HHS R01 DK067153NIDDK NIH HHS R01 DK074398NIDDK NIH HHS R01 DK075032NIDDK NIH HHS R01 DK083330NIH HHS 1DP2OD006486NIH HHS DP2 OD006486
6 · The paper itself

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.

Indexed as

AnimalsBody FluidsCell LineCellular Apoptosis Susceptibility ProteinCystic Fibrosis Transmembrane Conductance RegulatorDogsGastrointestinal TractGenes, RecessiveGreen Fluorescent ProteinsHomeostasisImmunoprecipitationMicroscopy, ConfocalMutationZebrafishZebrafish ProteinsCellular Apoptosis Susceptibility ProteinCFTR protein, zebrafishCystic Fibrosis Transmembrane Conductance RegulatorGreen Fluorescent ProteinsZebrafish Proteins

Identifiers

PMID20933420
PMCPMC2963654
OpenAlexW2058468930

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

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.