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Record W2068242642 · doi:10.1212/wnl.0b013e3181c677e1

INTERGENERATIONAL CONTRACTION OF THE CTG REPEATS IN 2 FAMILIES WITH MYOTONIC DYSTROPHY TYPE 1

2009· article· en· W2068242642 on OpenAlexaffabout
Jack Puymirat, Yves Giguère, Jean Mathieu, Jean‐Pierre Bouchard

Bibliographic record

VenueNeurology · 2009
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsHôpital de l'Enfant-Jésus
Fundersnot available
KeywordsMyotonic dystrophyMyotoniaContraction (grammar)Muscular dystrophyMedicineGeneticsTrinucleotide repeat expansionEndocrinologyInternal medicineBiologyGeneAllele

Abstract

fetched live from OpenAlex

Myotonic dystrophy type 1 (DM1) is an autosomal dominant multisystemic disorder caused by the expansion of an unstable CTG trinucleotide repeat at chromosome region 19q13.3.1 The number of CTG repeats is unstable in the abnormal range and usually increases in size in successive generations, in association with anticipation.2,3 A decrease in the CTG repeat size during transmission from parents to child can also occur in about 6.4% of transmissions, most frequently during paternal transmissions (10%).4 In the French-Canadian DM1 population, intergenerational contractions occur in about 7.4% of transmissions, all in cases of paternal transmission. We report here 2 French-Canadian DM1 families with paternal transmission, both originating from the Saguenay-Lac St-Jean, in which all affected children display CTG contractions. Although it was already reported that intergenerational contractions could be observed in several sibs in a same family, it was not noted whether this occurred in all affected sibs.4 Overall, these observations support the existence of a paternal factor that prevents CTG expansion. ### Case reports. The pedigree of the 2 families and Southern blot analysis are shown in the figure. Figure Family tree and Southern blot Pedigree of family (A, B). (C, D) Southern blot analysis. The DNA (3–5 μg) was …

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: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.119
Threshold uncertainty score0.237

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0020.001
Science and technology studies0.0040.001
Scholarly communication0.0010.000
Open science0.0010.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0030.000

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 head0.015
GPT teacher head0.244
Teacher spread0.230 · 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 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

Citations15
Published2009
Admission routes2
Has abstractyes

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