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Record W2003647187 · doi:10.1073/pnas.1010334108

Deletion of the ubiquitin ligase Nedd4L in lung epithelia causes cystic fibrosis-like disease

2011· article· en· W2003647187 on OpenAlexafffund
Toshihiro Kimura, Hiroshi Kawabe, Chong Jiang, Wenbo Zhang, Yun‐Yan Xiang, Chen Lu, Michael W. Salter, Nils Brose, Wei‐Yang Lu, Daniela Rotin

Bibliographic record

VenueProceedings of the National Academy of Sciences · 2011
Typearticle
Languageen
FieldMedicine
TopicCystic Fibrosis Research Advances
Canadian institutionsWestern UniversitySickKids FoundationHospital for Sick ChildrenUniversity of Toronto
FundersCanadian Institutes of Health ResearchCincinnati Children's Hospital Medical CenterCystic Fibrosis FoundationUniversity of TorontoMax-Planck-GesellschaftDeutsche ForschungsgemeinschaftJapan Society for the Promotion of ScienceMochida Memorial Foundation for Medical and Pharmaceutical ResearchHoward Hughes Medical Institute
KeywordsEpithelial sodium channelCystic fibrosisCystic fibrosis transmembrane conductance regulatorUbiquitin ligaseLungAmilorideFibrosisIdiopathic pulmonary fibrosisKnockout mouseUbiquitinBleomycinBiologyPathologyEndocrinologyInternal medicineMedicineChemistryBiochemistry

Abstract

fetched live from OpenAlex

Cystic fibrosis is caused by impaired ion transport due to mutated cystic fibrosis transmembrane conductance regulator, accompanied by elevated activity of the amiloride-sensitive epithelial Na(+) channel (ENaC). Here we show that knockout of the ubiquitin ligase Nedd4L (Nedd4-2) specifically in lung epithelia (surfactant protein C-expressing type II and Clara cells) causes cystic fibrosis-like lung disease, with airway mucus obstruction, goblet cell hyperplasia, massive inflammation, fibrosis, and death by three weeks of age. These effects of Nedd4L loss are likely caused by enhanced ENaC function, as reflected by increased ENaC protein levels, increased lung dryness at birth, amiloride-sensitive dehydration of lung explants, and elevated ENaC currents in primary alveolar type II cells analyzed by patch clamp recordings. Moreover, the lung defects were rescued with administration of amiloride into the lungs of young knockout pups via nasal instillation. Our results therefore suggest that the ubiquitin ligase Nedd4L can suppress the onset of cystic fibrosis symptoms by inhibiting ENaC in lung epithelia.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.003
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.411
Threshold uncertainty score0.516

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.043
GPT teacher head0.327
Teacher spread0.285 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations115
Published2011
Admission routes2
Has abstractyes

Explore more

Same venueProceedings of the National Academy of SciencesSame topicCystic Fibrosis Research AdvancesFrench-language works237,207