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Record W4392242077 · doi:10.26508/lsa.202302181

A comparative study of structural variant calling in WGS from Alzheimer’s disease families

2024· article· en· W4392242077 on OpenAlexafffund
John Malamon, John J. Farrell, Beth A. Dombroski, Gautami Das, Jessica Way, Amanda B Kuzma, Otto Valladares, Yuk Yee Leung, Allison J. Scanlon, Irving Barrera, Jack Brehony, Kim C. Worley, Nancy R. Zhang, Li‐San Wang, Lindsay A. Farrer, Gerard D. Schellenberg, Wan‐Ping Lee, Badri N. Vardarajan

Bibliographic record

VenueLife Science Alliance · 2024
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsInstitute of Aging
FundersU.S. National Library of MedicineNational Institute of Neurological Disorders and StrokeNational Institute on Deafness and Other Communication DisordersNational Heart, Lung, and Blood InstituteUniformed Services University of the Health SciencesNational Institutes of HealthÖsterreichische ForschungsförderungsgesellschaftNational Institute on AgingMedizinische Universität GrazBroad InstituteOesterreichische NationalbankNederlandse Organisatie voor Wetenschappelijk OnderzoekNational Alzheimer's Coordinating CenterErasmus Medisch CentrumAustrian Science FundZonMwVanderbilt UniversityEU Joint Programme – Neurodegenerative Disease ResearchEuropean CommissionUniversity of PennsylvaniaNational Human Genome Research InstituteRussian Foundation for Basic ResearchUniversity of MiamiUniversity of TorontoCase Western Reserve UniversityKarl-Franzens-Universität GrazU.S. Department of Health and Human Services
KeywordsSanger sequencingIndelGenotypingBiologyGeneticsComputational biologyGenomeWhole genome sequencingBreakpointDNA sequencingSingle-nucleotide polymorphismGeneGenotypeChromosome

Abstract

fetched live from OpenAlex

Detecting structural variants (SVs) in whole-genome sequencing poses significant challenges. We present a protocol for variant calling, merging, genotyping, sensitivity analysis, and laboratory validation for generating a high-quality SV call set in whole-genome sequencing from the Alzheimer's Disease Sequencing Project comprising 578 individuals from 111 families. Employing two complementary pipelines, Scalpel and Parliament, for SV/indel calling, we assessed sensitivity through sample replicates (N = 9) with in silico variant spike-ins. We developed a novel metric, D-score, to evaluate caller specificity for deletions. The accuracy of deletions was evaluated by Sanger sequencing. We generated a high-quality call set of 152,301 deletions of diverse sizes. Sanger sequencing validated 114 of 146 detected deletions (78.1%). Scalpel excelled in accuracy for deletions ≤100 bp, whereas Parliament was optimal for deletions >900 bp. Overall, 83.0% and 72.5% of calls by Scalpel and Parliament were validated, respectively, including all 11 deletions called by both Parliament and Scalpel between 101 and 900 bp. Our flexible protocol successfully generated a high-quality deletion call set and a truth set of Sanger sequencing-validated deletions with precise breakpoints spanning 1-17,000 bp.

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 score0.655
Threshold uncertainty score0.372

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 head0.032
GPT teacher head0.320
Teacher spread0.288 · 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

Citations6
Published2024
Admission routes2
Has abstractyes

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