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Record W2567630940 · doi:10.1016/s1525-0016(16)32990-2

181. Optimization of In Vivo Delivery of Baculovirus Gene Therapy Vectors for the Treatment of Alpha-1 Antitrypsin Deficiency

2016· article· en· W2567630940 on OpenAlexaff
Laura vanLieshout, Jondavid de Jong, Jacob P. vanVloten, Sarah K. Wootton

Bibliographic record

VenueMolecular Therapy · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicViral Infectious Diseases and Gene Expression in Insects
Canadian institutionsUniversity of Guelph
Fundersnot available
KeywordsGenetic enhancementGene deliveryNeutrophil elastaseAlpha 1-antitrypsin deficiencyImmunologyBiologyLungIn vivoVector (molecular biology)ElastaseReporter geneMedicineGeneInflammationGene expressionRecombinant DNAGenetics

Abstract

fetched live from OpenAlex

Alpha-1 antitrypsin deficiency (AATD) is a single-gene disorder commonly associated with adult onset lung diseases, including emphysema, chronic obstructive pulmonary disease and airway inflammation. Alpha-1 antitrypsin (AAT) is produced primarily in hepatocytes and is subsequently secreted into the bloodstream and travels to the lung where it inhibits neutrophil elastase. Unregulated neutrophil elastase activity in individuals with AATD causes degradation of various components of the extracellular matrix producing damage to alveolar and airway epithelial cells. Furthermore, mutated AAT folds incorrectly causing intracellular aggregation within hepatocytes, which can result in liver damage in addition to pulmonary symptoms. The development of gene therapy strategies has been a popular approach for the treatment of AATD due to monogenic nature of this genetic disease. Recently, baculovirus based vectors have garnered attention for possible application in gene therapy due to multiple factors. Baculoviruses can tolerate large gene insertions (>38kb), are capable of transducing mammalian cells but not replicating or integrating into host chromosomes and finally, humans lack pre-existing immunity to these insect viruses. Although there are many characteristics that make baculovirus vectors attractive for use in gene therapy, there is little in vivo data on bio-distribution and tissue tropism in mammalian models. As AATD is a disease affecting both the liver and the lung, we examined several delivery routes of a baculovirus vector expressing the human placental alkaline phosphatase reporter gene in C57BL/6 mice. To target the lung we investigated intranasal and intratracheal administration and for liver delivery intravenous, intrahepatic and intraportal vein injections were utilized. Lung delivery of wild type baculovirus yielded little transduction, however intrahepatic injection resulted in moderate liver transduction as well as unexpected spread to other tissues. Different formulations of the baculovirus vector were examined for pulmonary delivery including a variety of viscoeleastic gels. Additionally, pharmacological agents such as clodronate liposomes to deplete macrophages and cobra venom factor to ablate complement activity were examined in an effort to bolster transduction.

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)0.000
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: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.022
Threshold uncertainty score0.319

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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)0.0000.000

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 head0.014
GPT teacher head0.264
Teacher spread0.250 · 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

Citations0
Published2016
Admission routes1
Has abstractyes

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