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Record W4401938199 · doi:10.1101/2024.08.26.609771

Distinct pathogenic influence of anti-HMGCR+ and anti-SRP+ immune-mediated necrotizing myopathy autoantibodies on engineered muscle function

2024· preprint· en· W4401938199 on OpenAlexaff
Heta Lad, Ernest Myguel Esteban, Yekaterina Tiper, Alexandrine Mahoudeau, Zhuoye Xie, B. Tendrel, Yves Allenbach, Olivier Benvéniste, Penney M. Gilbert

Bibliographic record

VenuebioRxiv (Cold Spring Harbor Laboratory) · 2024
Typepreprint
Languageen
FieldMedicine
TopicInflammatory Myopathies and Dermatomyositis
Canadian institutionsUniversity of Toronto
Fundersnot available
KeywordsSarcomereAutoantibodySkeletal muscleImmune systemMyocytePathogenesisContraction (grammar)MyopathyImmunologyMedicineAntibodyInternal medicineBiology

Abstract

fetched live from OpenAlex

Abstract Immune-mediated necrotizing myopathy (IMNM) is a subgroup of idiopathic inflammatory myopathies associated with anti-signal recognition particle (SRP) or anti-3-hydroxy-3-methylglutaryl-CoA reductase (HMGCR) autoantibodies. However, the demonstration of a direct pathogenic effect of IMNM patient autoantibodies on skeletal muscle contractile force, independent of the downstream activation of the complement pathway, has yet to be reported. Thus, the goal of this study was to leverage a custom 3D-human skeletal muscle microtissue (hMMT) culture platform, that enables muscle cell contractile apparatus maturation and the analysis of contractile function, to evaluate the direct effect of total immunoglobulins (IgGs) isolated from IMNM patients with amplification of anti-SRP + or anti-HMGCR + autoantibodies. hMMTs capable of force generation were treated with total IgGs, isolated from 3 SRP+ and 3 HMGCR+ patients plasma, and delivered in complement inactivated media for 4 days. hMMT health was then evaluated by quantifying the peak force and contraction kinetics in response to electrical field stimulation and by performing histological analysis of sarcomere and myotube structures. Treating hMMTs with total IgGs from anti-HMGCR + patients resulted in a decline in tetanus contractile force, though sarcomere Z-line architecture analysis revealed no significant influences on sarcomere organization. hMMT treatment with total IgGs from anti-SRP + patients induced muscle atrophy, observed via significantly smaller myotube diameter, but this did not translate to a decline in contractile function. This study demonstrates that anti-SRP and anti-HMGCR autoantibodies exert direct, but distinct influences on IMNM-associated skeletal muscle pathogenesis, which may inform IMNM therapy development.

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)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
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 score0.879
Threshold uncertainty score0.999

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.001
Research integrity0.0010.001
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 head0.007
GPT teacher head0.206
Teacher spread0.199 · 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.

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

Citations3
Published2024
Admission routes1
Has abstractyes

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