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The amyotrophic lateral sclerosis (ALS) and frontotemporal dementia (FTD)‐associated C9ORF72 r(GGGGCC)n hexanucleotide repeat forms extremely stable uni‐and multi‐molecular RNA G‐quadruplex structures

2013· article· en· W3171318868 on OpenAlexaff
Kaalak Reddy, Bita Zamiri, Sabrina Y. Stanley, Robert B. Macgregor, Christopher E. Pearson

Bibliographic record

VenueThe FASEB Journal · 2013
Typearticle
Languageen
FieldMedicine
TopicNeurogenetic and Muscular Disorders Research
Canadian institutionsOntario Drug Policy Research NetworkHospital for Sick ChildrenUniversity of Toronto
Fundersnot available
KeywordsC9orf72Frontotemporal dementiaRNA splicingAmyotrophic lateral sclerosisMyotonic dystrophyBiologyAlternative splicingGeneTrinucleotide repeat expansionGeneticsMolecular biologyRNAExonMedicineDiseaseDementiaPathology

Abstract

fetched live from OpenAlex

G‐rich nucleic acids in promoters, telomere repeats, immunoglobulin class switch motifs, and fragile X repeats can form G‐quadruplex structures. Quadruplexes can affect promoter activity, splicing and neurite mRNA targeting. Amyotrophic lateral sclerosis (ALS), commonly referred to as Lou Gehrig's disease, and frontotemporal dementia (FTD) were recently shown to be caused by expansion of a G‐rich (GGGGCC)•(CCCCGG) hexanucleotide repeat in the C9ORF72 gene and these mutant transcripts can aggregate in nuclear foci possibly sequestering repeat‐binding proteins. Here we demonstrate that the (GGGGCC)n RNA forms extremely stable uni‐ and multi‐molecular parallel G‐quadruplex structures, whose formation is influenced by repeat length. Furthermore, MBNL1, a structure‐specific splicing factor that is sequestered in myotonic dystrophy type 1 patients by binding to expanded CUG RNA repeats, does not bind the ALS‐FTD RNA hexanucleotide repeat, while the ASF/SF2 splicing factor can bind the repeat. These observations may shed light on the normal (GGGGCC)•(CCCCGG) repeat function in C9ORF72, its interacting partners, and upon pathogenic roles the expanded repeat may have in ALS‐FTD.

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)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.005

Distilled classifier scores by category (both heads)

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 head0.028
GPT teacher head0.257
Teacher spread0.229 · 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 designBench or experimental
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

Citations0
Published2013
Admission routes1
Has abstractyes

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