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Record W2951968424 · doi:10.1002/acn3.582

Whole‐exome sequencing in 20,197 persons for rare variants in Alzheimer's disease

2018· article· en· W2951968424 on OpenAlexfundno aff
Neha Raghavan, Adam M. Brickman, Howard Andrews, Jennifer J. Manly, Nicole Schupf, Rafael Lantigua, Charles J. Wolock, Sitharthan Kamalakaran, Slavé Petrovski, Giuseppe Tosto, Badri N. Vardarajan, David B. Goldstein, Richard Mayeux

Bibliographic record

VenueAnnals of Clinical and Translational Neurology · 2018
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsnot available
FundersU.S. National Library of MedicineNational Institute of Neurological Disorders and StrokeNational Center for Advancing Translational SciencesNational Human Genome Research InstituteNational Institute of Mental HealthNational Heart, Lung, and Blood InstituteNational Institute on AgingNational Institutes of HealthNational Association for Colitis and Crohn's DiseaseUniversity of TorontoCase Western Reserve UniversityNational Institute of Child Health and Human DevelopmentNational Alzheimer's Coordinating CenterBaylor College of MedicineUniversity of MiamiMuscular Dystrophy AssociationUniversity of PennsylvaniaEllison Medical FoundationAmerican Academy of Child and Adolescent PsychiatryVanderbilt UniversityBiogenEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentBroad Institute
KeywordsExome sequencingLoss functionDiseaseMedicineExomeAlzheimer's diseaseGeneticsGeneBioinformaticsBiologyInternal medicineMutationPhenotype

Abstract

fetched live from OpenAlex

Abstract Objective The genetic bases of Alzheimer's disease remain uncertain. An international effort to fully articulate genetic risks and protective factors is underway with the hope of identifying potential therapeutic targets and preventive strategies. The goal here was to identify and characterize the frequency and impact of rare and ultra‐rare variants in Alzheimer's disease, using whole‐exome sequencing in 20,197 individuals. Methods We used a gene‐based collapsing analysis of loss‐of‐function ultra‐rare variants in a case–control study design with data from the Washington Heights‐Inwood Columbia Aging Project, the Alzheimer's Disease Sequencing Project and unrelated individuals from the Institute of Genomic Medicine at Columbia University. Results We identified 19 cases carrying extremely rare SORL 1 loss‐of‐function variants among a collection of 6,965 cases and a single loss‐of‐function variant among 13,252 controls ( P = 2.17 × 10 −8 ; OR : 36.2 [95% CI : 5.8–1493.0]). Age‐at‐onset was 7 years earlier for patients with SORL 1 qualifying variant compared with noncarriers. No other gene attained a study‐wide level of statistical significance, but multiple top‐ranked genes, including GRID 2 IP , WDR 76 and GRN , were among candidates for follow‐up studies. Interpretation This study implicates ultra‐rare, loss‐of‐function variants in SORL 1 as a significant genetic risk factor for Alzheimer's disease and provides a comprehensive dataset comparing the burden of rare variation in nearly all human genes in Alzheimer's disease cases and controls. This is the first investigation to establish a genome‐wide statistically significant association between multiple extremely rare loss‐of‐function variants in SORL 1 and Alzheimer's disease in a large whole‐exome study of unrelated cases and controls.

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)0.001
metaresearch head score (Gemma)0.001
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: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.009
Threshold uncertainty score0.018

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

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 head0.117
GPT teacher head0.388
Teacher spread0.271 · 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".

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Citations165
Published2018
Admission routes1
Has abstractyes

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