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Record W1585771370 · doi:10.1111/cge.12526

Characterization of a novel founder <i><scp>MSH6</scp></i> mutation causing Lynch syndrome in the French Canadian population

2014· article· en· W1585771370 on OpenAlexafffundabout
Ester Castellsagué, J. Liu, Alexandra Volenik, Sylvie Giroux, R. Gagné, Bruno Maranda, A. Roussel‐Jobin, Jean Latreille, Rachel Laframboise, Laura Palma, Lidia Kasprzak, Victoria Marcus, Magali Bréguet, Serge Nolet, Zaki El-Haffaf, Karlene Australie, Adrian Gologan, Olga Aleynikova, K. Oros‐Klein, Celia M.T. Greenwood, Anne‐Marie Mes‐Masson, Diane Provencher, Marc Tischkowitz, George Chong, François Rousseau, William D. Foulkes

Bibliographic record

VenueClinical Genetics · 2014
Typearticle
Languageen
FieldMedicine
TopicGenetic factors in colorectal cancer
Canadian institutionsMcGill University Health CentreCentre de Santé et de Services Sociaux de ChicoutimiUniversité de MontréalUniversité LavalCentre hospitalier de l'Université LavalCentre Hospitalier de l’Université de MontréalMcGill UniversityThe Quebec Population Health Research NetworkUniversité de SherbrookeUniversité du Québec à MontréalJewish General Hospital
FundersCanadian Institutes of Health Research
KeywordsFounder effectLynch syndromeGeneticsMutationMSH6BiologyPopulationMedicineHaplotypeGeneAlleleDNA mismatch repairDNA repairGermline mutation

Abstract

fetched live from OpenAlex

We identified an MSH6 mutation (c.10C>T, p.Gln4*) causing Lynch syndrome (LS) in 11 French Canadian (FC) families from the Canadian province of Quebec. We aimed to investigate the molecular and clinical implications of this mutation among FC carriers and to assess its putative founder origin. We studied 11 probands and 27 family members. Additionally 6433 newborns, 187 colorectal cancer (CRC) cases, 381 endometrial cancer (EC) cases and 179 additional controls, all of them from Quebec, were used. Found in approximately 1 of 400 newborns, the mutation is one of the most common LS mutations described. We have found that this mutation confers a greater risk for EC than for CRC, both in the 11 studied families and in the unselected cases: EC [odds ratio (OR) = 7.5, p < 0.0001] and CRC (OR = 2.2, p = 0.46). Haplotype analyses showed that the mutation arose in a common ancestor, probably around 430-656 years ago, coinciding with the arrival of the first French settlers. Application of the results of this study could significantly improve the molecular testing and clinical management of LS families in Quebec.

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 categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.041
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
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 head0.061
GPT teacher head0.338
Teacher spread0.277 · 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

Citations22
Published2014
Admission routes3
Has abstractyes

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