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ATXN2 trinucleotide repeat length correlates with risk of ALS

2016· review· nl· W2550109145 on OpenAlexaff
William Sproviero, Aleksey Shatunov, Daniel Ståhl, Maryam Shoai, Wouter van Rheenen, Safa Al‐Sarraj, Peter M. Andersen, Nancy M. Bonini, F. L. Conforti, Philip Van Damme, Hussein Daoud, Maria del Mar Amador, Isabella Fogh, Monica Forzan, Ben Gaastra, Cinzia Gellera, Aaron D. Gitler, John Hardy, Pietro Fratta, Vincenzo La Bella, Isabelle Le Ber, Tim Van Langenhove, Serena Lattante, Yi‐Chung Lee, Andrea Malaspina, Vincent Meininger, Stéphanie Millecamps, Richard W. Orrell, Rosa Rademakers, Wim Robberecht, Guy A. Rouleau, Owen A. Ross, François Salachas, Katie Sidle, Bradley Smith, Bing‐Wen Soong, Gianni Sorarú, Giovanni Stévanin, Edor Kabashi, Claire Troakes, Christine Van Broeckhoven, Jan H. Veldink, Leonard H. van den Berg, Christopher E. Shaw, John Powell, Ammar Al‐Chalabi

Bibliographic record

VenueNeurobiology of Aging · 2016
Typereview
Languagenl
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsMcGill UniversityMontreal Neurological Institute and Hospital
FundersFonds Wetenschappelijk OnderzoekMedical Research CouncilBelgian Federal Science Policy OfficeKing's College LondonVlaamse regeringSouth London and Maudsley NHS Foundation TrustMotor Neurone Disease AssociationUniversiteit AntwerpenNational Institute of Neurological Disorders and StrokeNational Institute for Health and Care ResearchAssociation pour la Recherche sur la Sclérose Latérale Amyotrophique et autres Maladies du MotoneuroneWellcome Trust
KeywordsTrinucleotide repeat expansionAmyotrophic lateral sclerosisAlleleOdds ratioGeneticsBiologyConfidence intervalMedicineDiseaseInternal medicineGene

Abstract

fetched live from OpenAlex

We investigated a CAG trinucleotide repeat expansion in the ATXN2 gene in amyotrophic lateral sclerosis (ALS). Two new case-control studies, a British dataset of 1474 ALS cases and 567 controls, and a Dutch dataset of 1328 ALS cases and 691 controls were analyzed. In addition, to increase power, we systematically searched PubMed for case-control studies published after 1 August 2010 that investigated the association between ATXN2 intermediate repeats and ALS. We conducted a meta-analysis of the new and existing studies for the relative risks of ATXN2 intermediate repeat alleles of between 24 and 34 CAG trinucleotide repeats and ALS. There was an overall increased risk of ALS for those carrying intermediate sized trinucleotide repeat alleles (odds ratio 3.06 [95% confidence interval 2.37–3.94]; p = 6 × 10−18), with an exponential relationship between repeat length and ALS risk for alleles of 29–32 repeats (R2 = 0.91, p = 0.0002). No relationship was seen for repeat length and age of onset or survival. In contrast to trinucleotide repeat diseases, intermediate ATXN2 trinucleotide repeat expansion in ALS does not predict age of onset but does predict disease risk.

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)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow), Insufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Other design · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.688
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0040.001
Bibliometrics0.0010.001
Science and technology studies0.0000.002
Scholarly communication0.0000.000
Open science0.0010.001
Research integrity0.0010.002
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 head0.030
GPT teacher head0.309
Teacher spread0.278 · 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 designOther design
Domainnot available
GenreReview

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

Citations130
Published2016
Admission routes1
Has abstractyes

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