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Record W6987235452

Skeletal muscle adaptations to chronic exercise in a pre-clinical model of myotonic dystrophy type 1

2018· dissertation· en· W6987235452 on OpenAlexfundno aff

Bibliographic record

VenueMacSphere (McMaster University) · 2018
Typedissertation
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsnot available
FundersCanadian Institutes of Health ResearchNatural Sciences and Engineering Research Council of CanadaCanada Research Chairs
KeywordsSkeletal muscleMuscle atrophyElectromyographyTitinTSG101
DOInot available

Abstract

fetched live from OpenAlex

Myotonic dystrophy type 1 (DM1) is the second most common muscular dystrophy and most prevalent adult form. A microsatellite expansion comprised of CTG repetitions in the dystrophia myotonica protein kinase (DMPK) gene, DM1 is characterized by muscle weakness, wasting, and myotonia. The expanded nucleotide sequence of the DMPK mRNA results in the misregulation of important RNA-binding proteins (RNABPs), Muscleblind-like 1 (MBNL1) in particular. MBNL1 becomes trapped in myonuclei within the repeating CUG transcript, which reduces the RNABPs ability to process newly synthesized mRNAs that are important for the maintenance of healthy muscle function. Recent studies with DM1 participants demonstrate that exercise is safe, enjoyable, and elicits benefits in muscle strength and function. However, the molecular mechanisms of exercise adaptation in DM1 are largely unknown. Understanding the cellular processes that drive exercise-induced remodelling may assist in the discovery of effective lifestyle interventions to mitigate DM1. In this thesis, three groups of mice were utilized: i) sedentary DM1 animals (SED-DM1), ii) DM1 mice who volitionally exercised daily on a home cage running wheel (EX-DM1), and iii) sedentary healthy, wild-type mice (WT). EX-DM1 animals ran 5.6 km/day during the 7-week experimental time course, a volume of volitional physical activity that is lower than that observed in WT animals. Post-exercise functional tests demonstrated that chronic exercise significantly improved motor performance, muscle strength and endurance. Electromyography revealed that chronic exercise mitigated myotonia. At the cellular and molecular levels, we found that chronic physical activity attenuated RNA toxicity, liberated MBNL1 from myonuclear sequestration, and selectively normalized the spliceopathy of bridging integrator 1 and muscle-specific chloride channel mRNAs. Collectively, our data indicate that chronic exercise improves DM1 at the molecular, cellular and physiological levels.

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)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.009

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.000
Science and technology studies0.0010.001
Scholarly communication0.0010.000
Open science0.0010.000
Research integrity0.0010.002
Insufficient payload (model declined to judge)0.0030.001

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 head0.037
GPT teacher head0.275
Teacher spread0.238 · 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 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

Citations0
Published2018
Admission routes1
Has abstractyes

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