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Record W4406606072 · doi:10.1016/j.dscb.2025.100181

The evolving spectrum of LAMA2 related congenital muscular dystrophy (MDC1)-Case series and review of literature

2025· article· en· W4406606072 on OpenAlexaff
Rahul Sinha, Sonali Singh, Zulfikar Luhar, Ankit Kumar Meena, Arvinder Wander, Dharmesh Soneji

Bibliographic record

VenueBrain Disorders · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMuscle Physiology and Disorders
Canadian institutionsSickKids FoundationHospital for Sick ChildrenUniversity of Toronto
Fundersnot available
KeywordsSeries (stratigraphy)Muscular dystrophySpectrum (functional analysis)MedicineBiologyPhysicsInternal medicine

Abstract

fetched live from OpenAlex

• This case series provides varied spectrum in Laminin α2-related muscular dystrophy (LAMA2-MD) and to establish a well-characterized baseline cohort of patients for future studies. • The phenotypic spectrum of LAMA2 muscular dystrophy varies from severe early onset congenital muscular dystrophy (MCD1) to mild variant of late onset muscular dystrophy (LAMA2-MD). The MCD1is characterized by hypotonia, frequent respiratory and feeding issues with less commonly with epilepsy. • Neuroimaging findings aids in the diagnosis of MCD1. Involvement of corpus callosum (CC) in MCD1 may mimic the pattern of CC involvement in mitochondrial disorders. • Normal IQ, peripheral neuropathy, and borderline CPK values do not argue against the diagnosis of MDC1. Also, onset of clinical features in early infancy may not have severe outcome which is interesting in our study.

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)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.005
Threshold uncertainty score0.011

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0050.004
Science and technology studies0.0010.001
Scholarly communication0.0010.002
Open science0.0010.001
Research integrity0.0010.001
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 head0.002
GPT teacher head0.215
Teacher spread0.213 · 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 designCase report
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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