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Record W4413817845 · doi:10.1073/pnas.2506257122

TIGER: A tdTomato in vivo genome-editing reporter mouse for investigating precision-editor delivery approaches

2025· article· en· W4413817845 on OpenAlexfundno aff
Samuel W. Du, Grażyna Palczewska, Zhiqian Dong, Julie C. Lauterborn, Balasankara Reddy Kaipa, Alexander L. Yan, Rafał Hołubowicz, Siyoung Ha, Paul Chen, Christine M. Gall, Gulab S. Zode, David R. Liu, Krzysztof Palczewski

Bibliographic record

VenueProceedings of the National Academy of Sciences · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCRISPR and Genetic Engineering
Canadian institutionsnot available
FundersNational Institute of General Medical SciencesNational Eye InstituteHoward Hughes Medical InstituteNational Institute of Allergy and Infectious DiseasesNatural Sciences and Engineering Research Council of CanadaNational Institutes of HealthNational Human Genome Research InstituteSchool of Medicine, University of California, IrvineKnights Templar Eye FoundationUniversity of California, IrvineResearch to Prevent BlindnessEunice Kennedy Shriver National Institute of Child Health and Human Development
KeywordsGenome editingBiologyReporter geneIn vivoCell biologyGreen fluorescent proteinTransgeneEx vivoGenomeGene deliveryComputational biologyGeneticsGeneTransfectionGene expression

Abstract

fetched live from OpenAlex

In vivo genome editing has the potential to address many inherited and environmental disorders. However, a major hurdle for the clinical translation of genome editing is safe, efficient delivery to disease-relevant tissues. A modality-agnostic reporter animal model that facilitates rapid, precise, and quantifiable assessment of functional delivery and editing could greatly enhance the evaluation and translation of delivery technologies. Here, we present the development of the tdTomato in vivo genome-editing reporter (TIGER) mouse, a reporter strain that harbors an integrated and constitutively expressed mutated tdTomato gene in the Polr2a locus. The mutations (Q115X, Q357X) abolish fluorescence, but successful adenine base editing (ABE) or prime editing (PE) restores tdTomato fluorescence. This mouse model facilitates the tissue- and cell type–specific assessment of genome editing agent delivery. We describe several editing strategies validated in vitro and demonstrate efficient ABE and PE in vivo using viral and nonviral delivery vectors targeting four cell types within the mouse eye: the retinal pigment epithelium, photoreceptors, Müller glia, and the trabecular meshwork. We show direct editing characterization in the ocular tissues via in vivo and ex vivo two-photon confocal microscopy and verify the spectral and fluorescence lifetime properties of tdTomato reporter in other mouse tissues. Additionally, we demonstrate successful adeno-associated virus (AAV)-mediated PE of extraocular tissues, including hepatocytes, skeletal muscle, and brain neurons by intravenous injection. Thus, the TIGER mouse facilitates the direct development, comparison, and optimization of delivery platforms for efficient and productive ABE or PE broadly applicable in vivo across multiple tissues tested in this study.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.002
metaresearch head score (Gemma)0.001
Version: metacan-v3-hybrid-931329e0061cValidation 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 score0.012
Threshold uncertainty score0.039

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.001
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0040.001
Science and technology studies0.0010.001
Scholarly communication0.0010.002
Open science0.0020.001
Research integrity0.0030.004
Insufficient payload (model declined to judge)0.0120.004

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 head0.033
GPT teacher head0.316
Teacher spread0.283 · 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 source (direct Gemma or distilled Codex), 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

Citations4
Published2025
Admission routes1
Has abstractyes

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