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Record W4416758278 · doi:10.1038/s41514-025-00288-z

Perturbation of multiprotein complexes in skeletal muscle induces protective proteases in the CNS that degrade pathogenic proteins

2025· article· en· W4416758278 on OpenAlexfundno aff
Mamta Rai, Helen J. E. Baddeley, Chia-Lung Chuang, Anna Platt, Michelle Curley, David Finkelstein, Fabio Demontis

Bibliographic record

Venuenpj Aging · 2025
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsnot available
FundersNational Institute on AgingNational Institutes of HealthInstitute of GeneticsAmerican Lebanese Syrian Associated CharitiesSt. Jude Children's Research Hospital
KeywordsProteasesMultiprotein complexProteostasisDownregulation and upregulationSkeletal muscleMediatorSUMO protein

Abstract

fetched live from OpenAlex

Many cellular functions rely on multiprotein complexes and their stoichiometric assembly. Reducing the levels of individual complex components can perturb this process and induce corrective stress responses. In addition to local outcomes, cellular stress in one tissue can induce long-distance responses in other tissues. Here, we used muscle-targeted RNAi to examine the systemic stress responses induced by muscle-specific genetic perturbation of four distinct multiprotein complexes: the sarcomere, mitochondrial respiratory complex I, proteasome, and VCP (valosin-containing protein) complex. Muscle-specific disruption of these four complexes produced largely overlapping transcriptional adaptations in the central nervous system (CNS), and these responses were centered on the upregulation of many proteases and peptidases. Testing in a retinal model of Huntington's disease demonstrated that several stress-induced proteases limit the accumulation of huntingtin-polyQ aggregates during aging, indicating that these proteases protect from pathogenic proteins. We next examined whether the myokine Amyrel is a possible mediator of this stress-initiated muscle-to-CNS signaling because of its previously reported role in inducing protease expression. Consistent with this model, Amyrel expression was transcriptionally induced in muscle by perturbation of each of the four multiprotein complexes. Moreover, experimental upregulation of Amyrel in muscle reduced the amount of pathogenic huntingtin-polyQ aggregates in the retina. Taken together, these findings indicate that Amyrel and protective proteases improve CNS proteostasis following the perturbation of multiprotein complexes in skeletal muscle. Thus, this study provides insight into a muscle-to-CNS signaling axis that conveys information on the stress status of multiprotein complexes.

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: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.217
Threshold uncertainty score0.634

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 science0.0000.000
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 head0.044
GPT teacher head0.286
Teacher spread0.242 · 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

Citations2
Published2025
Admission routes1
Has abstractyes

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