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Record W4226145696 · doi:10.1038/s41525-022-00299-9

Genetic evidence supports the development of SLC26A9 targeting therapies for the treatment of lung disease

2022· article· en· W4226145696 on OpenAlexafffundabout
Jiafen Gong, Gengming He, Cheng Wang, Claire Bartlett, Naim Panjwani, Scott Mastromatteo, Lin Fan, Katherine Keenan, Julie Avolio, Anat Halevy, Michelle Shaw, Mohsen Esmaeili, Guillaume Côté-Maurais, Damien Adam, Stéphanie Bégin, Candice Bjornson, Mark Chilvers, Joe Reisman, April Price, Michael D. Parkins, Richard van Wylick, Yves Berthiaume, Lara Bilodeau, Dimas Mateos‐Corral, Daniel Hughes, Mary Jane Smith, Nancy Morrison, Janna Brusky, Elizabeth Tullis, Anne L. Stephenson, Bradley S. Quon, Pearce Wilcox, Winnie M. Leung, Melinda Solomon, Lei Sun, Emmanuelle Brochiero, Theo J. Moraes, Tanja Gonska, Félix Ratjen, Johanna M. Rommens, Lisa J. Strug

Bibliographic record

Venuenpj Genomic Medicine · 2022
Typearticle
Languageen
FieldMedicine
TopicCystic Fibrosis Research Advances
Canadian institutionsUniversity of Alberta HospitalAlberta Hospital EdmontonSt. Michael's HospitalInstitut universitaire de cardiologie et de pneumologie de QuébecMemorial University of NewfoundlandFoothills Medical CentreLondon Health Sciences CentreSt. Paul's HospitalIzaak Walton Killam Health CentreAlberta Children's HospitalUniversity of SaskatchewanHospital for Sick ChildrenUniversité de MontréalQueen Elizabeth II Health Sciences CentreChildren's Hospital of Eastern OntarioUniversité LavalBC Children's HospitalCentre Hospitalier de l’Université de MontréalKingston Health Sciences CentrePublic Health OntarioUniversity of Toronto
FundersHospital for Sick ChildrenNatural Sciences and Engineering Research Council of CanadaCystic Fibrosis CanadaGovernment of CanadaCanadian Institutes of Health ResearchGenome CanadaGovernment of OntarioMedical Research CouncilCystic Fibrosis Foundation TherapeuticsCystic Fibrosis Foundation
KeywordsMedicineCystic fibrosis transmembrane conductance regulatorCystic fibrosisCOPDPopulationSpirometryInternal medicineBiobankLungPharmacologyBioinformaticsOncologyBiologyEnvironmental health

Abstract

fetched live from OpenAlex

Abstract Over 400 variants in the cystic fibrosis (CF) transmembrane conductance regulator ( CFTR ) are CF-causing. CFTR modulators target variants to improve lung function, but marked variability in response exists and current therapies do not address all CF-causing variants highlighting unmet needs. Alternative epithelial ion channel/transporters such as SLC26A9 could compensate for CFTR dysfunction, providing therapeutic targets that may benefit all individuals with CF. We investigate the relationship between rs7512462, a marker of SLC26A9 activity, and lung function pre- and post-treatment with CFTR modulators in Canadian and US CF cohorts, in the general population, and in those with chronic obstructive pulmonary disease (COPD). Rs7512462 CC genotype is associated with greater lung function in CF individuals with minimal function variants (for which there are currently no approved therapies; p = 0.008); and for gating ( p = 0.033) and p.Phe508del/ p.Phe508del ( p = 0.006) genotypes upon treatment with CFTR modulators. In parallel, human nasal epithelia with CC and p.Phe508del/p.Phe508del after Ussing chamber analysis of a combination of approved and experimental modulator treatments show greater CFTR function ( p = 0.0022). Beyond CF, rs7512462 is associated with peak expiratory flow in a meta-analysis of the UK Biobank and Spirometa Consortium ( p = 2.74 × 10 −44 ) and provides p = 0.0891 in an analysis of COPD case-control status in the UK Biobank defined by spirometry. These findings support SLC26A9 as a therapeutic target to improve lung function for all people with CF and in individuals with other obstructive lung diseases.

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)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesInsufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.522
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

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 head0.042
GPT teacher head0.345
Teacher spread0.303 · 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.

Study designObservational
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

Citations17
Published2022
Admission routes3
Has abstractyes

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