MétaCan
Menu
Back to cohort
Record W2596924828 · doi:10.1002/humu.23195

Overstressed response to <i>EIF2S3</i> variants in MEHMO syndrome

2017· letter· en· W2596924828 on OpenAlexaff
Jacques L. Michaud

Bibliographic record

VenueHuman Mutation · 2017
Typeletter
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicRNA modifications and cancer
Canadian institutionsCentre Hospitalier Universitaire Sainte-JustineUniversité de Montréal
Fundersnot available
KeywordsBiologyIntegrated stress responseTranslation (biology)GeneticsMicrocephalyMutationContext (archaeology)Unfolded protein responseEukaryotic translationGeneMessenger RNA

Abstract

fetched live from OpenAlex

In response to diverse stress stimuli, eukaryotic cells activate a common adaptive pathway, termed the integrated stress response (ISR). The core event in this pathway is a decrease of the eukaryotic translation initiation factor 2 (eIF2), which plays a crucial role for the initiation of translation. Decrease of eIF2 in the context of the ISR leads to a decrease in global protein synthesis and the induction of specific genes. Paradoxically, although the ISR primarily promotes cellular recovery, exposure to severe stress such as fever or head trauma can drive signaling toward cell death. Skopkova et al. (Hum Mutat 38: 409–425, 2017) report in this issue that disruption of EIF2S3, which codes for the subunit γ of eIF2, causes MEHMO syndrome. This disorder is characterized by severe intellectual disability, epilepsy, hypogonadism, microcephaly and obesity. The authors identified X-linked recessive variants in EIF2S3 in four families with MEHMO syndrome, three of which carry the same mutation, possibly because they share a common ancestor. Elegant studies performed in yeast showed that this variant decreases EIF2S3 function. Interestingly, studies in fibroblasts from one of the patients further suggest that the mutation in EIF2S3 is associated with an activation of ISR. MEHMO syndrome represents a new addition to a growing group of disorders that are associated with the function or availability of eIF2, causing dysregulation of translation. These disorders are characterized by a general impairment of protein synthesis and increased sensitivity in states of elevated metabolic requirements, resulting in apoptosis and the occurrence of episodes of rapid clinical deterioration following major stresses (as documented in some patients with MEHMO syndrome). The work of Skopkova et al. thus offers an important new perspective on this emerging chapter of medicine.

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)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.169
Threshold uncertainty score0.894

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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 head0.021
GPT teacher head0.296
Teacher spread0.275 · 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 designNot applicable
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

Citations1
Published2017
Admission routes1
Has abstractyes

Explore more

Same venueHuman MutationSame topicRNA modifications and cancerFrench-language works237,207