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Record W2097287826 · doi:10.1212/wnl.0b013e318219faf6

Lafora bodies in skeletal muscle are fiber type specific

2011· article· en· W2097287826 on OpenAlexafffund
Julie Turnbull, Jean‐Marie Girard, Nela Pencea, Xiaochu Zhao, Terry E. Graham, Pengyu Wang, Cameron Ackerley, Berge A. Minassian

Bibliographic record

VenueNeurology · 2011
Typearticle
Languageen
FieldMedicine
TopicGlycogen Storage Diseases and Myoclonus
Canadian institutionsUniversity of Toronto
FundersCanadian Institutes of Health Research
KeywordsLafora diseaseGlycogenChemistryBiochemistryProgressive myoclonus epilepsyGlycogen synthaseGlycogen phosphorylaseNeurodegenerationGlycogen branching enzymePhosphorylationBiologyEndocrinologyInternal medicinePhosphataseGeneMedicine

Abstract

fetched live from OpenAlex

Glycogen is the largest soluble cytosolic macromolecule, containing up to 55,000 glucoses per molecule. It is formed by glycogen synthase (GS) and branching enzyme (BE), which acting coordinately lead to branching after every sixth glucose and ultimately to a spherical shape that allows solubility. Polyglucosans are malformed glycogen molecules containing much less branching. They precipitate and accumulate into polyglucosan bodies (PB). PB characterize adult polyglucosan body disease (APBD) and Lafora disease (LD). In APBD, PB form in and often obstruct axons. APBD is an axonopathy with upper and lower motor neuron signs and no epilepsy. In LD, PB, called Lafora bodies (LB), occupy neuronal perikarya and dendrites. LD is a progressive myoclonus epilepsy, with no long tract or peripheral nerve deficits. APBD is caused by BE deficiency. LD is caused by mutations in the EPM2A and EPM2B genes encoding respectively the laforin phosphatase and the malin ubiquitin E3 ligase, which regulates laforin. In LD, glycogen becomes progressively phosphorylated, a pathologic process normally prevented by laforin. The charged phosphates unfold glycogen, expose its hydrophobic regions, and lead it to precipitate. GS precipitates with glycogen, but BE does not. Subsequent extension by GS unchecked by branching would convert the glycogen to polyglucosan.1,2 The next step in LD research is to understand the basis of the progressive phosphorylation. What is known is …

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 categoriesInsufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.430
Threshold uncertainty score0.999

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)0.0020.001

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 head0.036
GPT teacher head0.250
Teacher spread0.215 · 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 designObservational
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

Citations9
Published2011
Admission routes2
Has abstractyes

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