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Record W3047049660 · doi:10.1038/s41467-020-17577-8

A lung tropic AAV vector improves survival in a mouse model of surfactant B deficiency

2020· article· en· W3047049660 on OpenAlexafffund
Martin H. Kang, Laura P. van Lieshout, Liqun Xu, Jakob M. Domm, Arul Vadivel, Laurent Renesme, Christian Mühlfeld, Maria Hurskainen, Ivana Mižíková, Yanlong Pei, Jacob P. van Vloten, Sylvia P. Thomas, Claudia Milazzo, Chanèle Cyr-Depauw, Jeffrey A. Whitsett, Lawrence M. Nogee, Sarah K. Wootton, Bernard Thébaud

Bibliographic record

VenueNature Communications · 2020
Typearticle
Languageen
FieldMedicine
TopicNeonatal Respiratory Health Research
Canadian institutionsUniversity of GuelphChildren's Hospital of Eastern OntarioOttawa HospitalUniversity of Ottawa
FundersCanadian Institutes of Health ResearchDeutsche ForschungsgemeinschaftNational Heart, Lung, and Blood InstituteLloyd's Tercentenary Research FoundationNational Institutes of HealthGovernment of CanadaEuropean Respiratory SocietyUniversity of Ottawa
KeywordsLungGenetic enhancementPulmonary surfactantRespiratory distressBiologyVector (molecular biology)Surfactant protein CImmunologyFlow cytometryTransgeneHomeostasisMedicinePathologyGeneCell biologyInternal medicineGenetics

Abstract

fetched live from OpenAlex

Surfactant protein B (SP-B) deficiency is an autosomal recessive disorder that impairs surfactant homeostasis and manifests as lethal respiratory distress. A compelling argument exists for gene therapy to treat this disease, as de novo protein synthesis of SP-B in alveolar type 2 epithelial cells is required for proper surfactant production. Here we report a rationally designed adeno-associated virus (AAV) 6 capsid that demonstrates efficiency in lung epithelial cell transduction based on imaging and flow cytometry analysis. Intratracheal administration of this vector delivering murine or human proSFTPB cDNA into SP-B deficient mice restores surfactant homeostasis, prevents lung injury, and improves lung physiology. Untreated SP-B deficient mice develop fatal respiratory distress within two days. Gene therapy results in an improvement in median survival to greater than 200 days. This vector also transduces human lung tissue, demonstrating its potential for clinical translation against this lethal disease.

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)0.000
metaresearch head score (Gemma)0.001
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 score0.890
Threshold uncertainty score0.696

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0000.002
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 head0.084
GPT teacher head0.396
Teacher spread0.311 · 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

Citations76
Published2020
Admission routes2
Has abstractyes

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