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Record W4414899992 · doi:10.1126/scisignal.adx3087

Phosphorylation of RYR1 at Ser <sup>2902</sup> decreases Ca <sup>2+</sup> leak in skeletal muscle and susceptibility to malignant hyperthermia and heat stroke

2025· article· en· W4414899992 on OpenAlexaff
Rachel Sue Zhen Yee, Chang Seok Lee, Ting Chang, Sung Yun Jung, Omar Yousif, Courtney L. Cavazos, John Colyer, Filip Van Petegem, George G. Rodney, Susan L. Hamilton

Bibliographic record

VenueScience Signaling · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicIon channel regulation and function
Canadian institutionsUniversity of British Columbia
FundersNational Institute of Diabetes and Digestive and Kidney DiseasesNational Cancer InstituteNational Institute of Arthritis and Musculoskeletal and Skin DiseasesNational Human Genome Research InstituteNational Institutes of Health
KeywordsRYR1Malignant hyperthermiaSkeletal muscleRyanodine receptorPhosphorylationEndoplasmic reticulumPoint mutation

Abstract

fetched live from OpenAlex

Ryanodine receptor 1 (RYR1) is the sarcoplasmic reticulum (SR) Ca 2+ release channel required for both skeletal muscle contraction and Ca 2+ leak. Mutations in RYR1 cause malignant hyperthermia susceptibility (MHS) and enhanced sensitivity to heat stroke (ESHS), which can result in death due to excessive skeletal muscle thermogenesis upon exposure to volatile anesthetics or heat. Here, we investigated the molecular and physiological functions of phosphorylation of RYR1 at Ser 2902 by the kinase striated muscle preferentially expressed protein (SPEG). Muscle from SPEG-deficient mice expressing RYR1 with a Ser 2902 →Asp 2902 (S2902D) point mutation to mimic phosphorylation by SPEG showed decreased SR Ca 2+ sparks. Muscle from mice homozygous for the S2902D point mutation had reduced SR Ca 2+ transients and small changes in force generation but overall mild phenotypic changes. YS mice, which are heterozygous for a Tyr 524 →Ser 524 point mutation in RYR1, show increased Ca 2+ leak and are a model of MHS and ESHS. Crossing YS mice with S2902D mice led to decreased SR Ca 2+ leak and desensitized the mice to both volatile anesthetics and heat. Thus, SPEG inhibits SR Ca 2+ leak in skeletal muscle by phosphorylating Ser 2902 on RYR1, and mutation of Ser 2902 to Asp 2902 to mimic this phosphorylation event rescues YS mice from heat-induced death.

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 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 score0.107
Threshold uncertainty score0.499

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
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 head0.009
GPT teacher head0.245
Teacher spread0.236 · 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
Published2025
Admission routes1
Has abstractyes

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