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Record W2028888671 · doi:10.1093/jnci/dju384

Germline Mutations in Shelterin Complex Genes Are Associated With Familial Glioma

2014· article· en· W2028888671 on OpenAlexaff
Matthew N. Bainbridge, Georgina Armstrong, M. Monica Gramatges, Alison A. Bertuch, Shalini N. Jhangiani, HarshaVardhan Doddapaneni, Lora Lewis, Joseph Tombrello, Spyros Tsavachidis, Yanhong Liu, Ali Jalali, Sharon E. Plon, Ching C. Lau, D. Williams Parsons, Elizabeth B. Claus, Jill S. Barnholtz‐Sloan, Dora Il’yasova, Joellen M. Schildkraut, Francis Ali‐Osman, Siegal Sadetzki, Christoffer Johansen, Richard S. Houlston, Robert B. Jenkins, Daniel H. Lachance, Sara H. Olson, Jonine L. Bernstein, Ryan Merrell, Margaret Wrensch, Kyle M. Walsh, Faith G. Davis, Rose Lai, Sanjay Shete, Kenneth Aldape, Christopher I. Amos, Patricia A. Thompson, Donna M. Muzny, Richard A. Gibbs, Beatrice Melin, Melissa L. Bondy

Bibliographic record

VenueJNCI Journal of the National Cancer Institute · 2014
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicEpigenetics and DNA Methylation
Canadian institutionsUniversity of Alberta
FundersNational Human Genome Research InstituteSt. Baldrick's FoundationNational Brain Tumor SocietyU.S. Public Health ServiceNational Cancer InstituteNational Institutes of HealthAmerican Brain Tumor Association
KeywordsGliomaShelterinExome sequencingGeneticsBiologyOligodendrogliomaGermlineGermline mutationMutationExomeGeneCancer researchAstrocytomaDNA-binding proteinTranscription factor

Abstract

fetched live from OpenAlex

Gliomas are the most common brain tumor, with several histological subtypes of various malignancy grade. The genetic contribution to familial glioma is not well understood. Using whole exome sequencing of 90 individuals from 55 families, we identified two families with mutations in POT1 (p.G95C, p.E450X), a member of the telomere shelterin complex, shared by both affected individuals in each family and predicted to impact DNA binding and TPP1 binding, respectively. Validation in a separate cohort of 264 individuals from 246 families identified an additional mutation in POT1 (p.D617Efs), also predicted to disrupt TPP1 binding. All families with POT1 mutations had affected members with oligodendroglioma, a specific subtype of glioma more sensitive to irradiation. These findings are important for understanding the origin of glioma and could have importance for the future diagnostics and treatment of glioma.

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 categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.774
Threshold uncertainty score0.229

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)0.0000.000

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 head0.043
GPT teacher head0.319
Teacher spread0.276 · 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.

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

Citations229
Published2014
Admission routes1
Has abstractyes

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