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Record W4382345469 · doi:10.1016/j.xgen.2023.100316

Genome-wide structural variant analysis identifies risk loci for non-Alzheimer’s dementias

2023· article· en· W4382345469 on OpenAlexaff
Karri Kaivola, Ruth Chia, Jinhui Ding, Memoona Rasheed, Masashi Fujita, Vilas Menon, Ronald L. Walton, Ryan L. Collins, Kimberley J. Billingsley, Harrison Brand, Michael E. Talkowski, Xuefang Zhao, Ramita Dewan, Ali Stark, Anindita Ray, Sultana Solaiman, Pilar Álvarez Jerez, Laksh Malik, Ted M. Dawson, Liana S. Rosenthal, Marilyn S. Albert, Olga Pletnikova, Juan C. Troncoso, Mario Masellis, Julia Keith, Sandra E. Black, Luigi Ferrucci, Susan M. Resnick, Toshiko Tanaka, Anthony R. Soltis, Coralie Viollet, Gauthaman Sukumar, Camille Alba, Nathaniel M. Lott, Elisa McGrath Martinez, Meila Tuck, Jatinder Singh, Dagmar Bačíková, Xijun Zhang, Daniel N. Hupalo, Adelani Adeleye, Matthew D. Wilkerson, Harvey B. Pollard, Clifton L. Dalgard, Ziv Gan‐Or, Ekaterina Rogaeva, Alexis Brice, Suzanne Lesage, Georgia Xiromerisiou, Andrea Calvo, Antonio Canosa, Adriano Chiò, Giancarlo Logroscino, Gabriele Mora, Reijko Krüger, Patrick May, Daniel Alcolea, Jordi Clarimón, Juan Fortea, Isabel González Aramburu, Jon Infante, Carmen Lage, Alberto Lleó, Pau Pástor, Pascual Sánchez‐Juan, Francesca Brett, Dag Aarsland, Safa Al‐Sarraj, Johannes Attems, Steve Gentleman, John Hardy, Angela Hodges, Seth Love, Ian G. McKeith, Christopher M. Morris, Huw R. Morris, Stuart Pickering‐Brown, Alan Thomas, Claire Troakes, Matthew J. Barrett, Thomas G. Beach, Lynn M. Bekris, David A. Bennett, Bradley F. Boeve, Dennis W. Dickson, Kelley Faber, Margaret E. Flanagan, Tatiana Foroud, Alison Goate, David S. Goldstein, Horacio Kaufmann, Walter A. Kukull, James B. Leverenz, Grisel Lopez, Qinwen Mao, Eliezer Masliah, Edwin S. Monuki, Kathy L. Newell, Jose‐Alberto Palma, Matthew Perkins, Alan E. Renton, Owen A. Ross, Clemens R. Scherzer, Vikram G. Shakkottai, Ellen Sidransky, Nahid Tayebi, Eric Topol, Ali Torkamani, Randy Woltjer, Zbigniew K. Wszołek, Sonja W. Scholz, Robert H. Baloh, Robert Bowser, James R. Broach, William Camu, Johnathan Cooper‐Knock, Carsten Drepper, Vivian E. Drory, Travis Dunckley, Eva L. Feldman, Pietro Fratta, Glenn S. Gerhard, Summer Gibson, Jonathan D. Glass, Matthew B. Harms, Terry Heiman‐Patterson, Lilja Jansson, Janine Kirby, Justin Kwan, Hannu Laaksovirta, John E. Landers, Francesco Landi, Isabelle Le Ber, Serge Lumbroso, Daniel J. MacGowan, Nicholas J. Maragakis, Kévin Mouzat, Liisa Myllykangas, Richard W. Orrell, Lyle W. Ostrow, Roger Pamphlett, Erik P. Pioro, Stefan M. Pulst, John Ravits, Wim Robberecht, Jeffrey D. Rothstein, Michael Sendtner, Pamela J. Shaw, Katie Sidle, Zachary Simmons, Thor D. Stein, David J. Stone, Pentti J. Tienari, Bryan J. Traynor, Miko Valori, Philip Van Damme, Vivianna M. Van Deerlin, Ludo Van Den Bosch, Lorne Zinman, Letizia Mazzini, Sandra D’Alfonso, Cristina Moglia, Philip L. De Jager

Bibliographic record

VenueCell Genomics · 2023
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsCanada Research ChairsSunnybrook Health Science CentreHeart and Stroke FoundationUniversity of TorontoUniversity of New BrunswickHealth Sciences Centre
FundersNational Institute of Neurological Disorders and StrokeNational Human Genome Research InstituteNational Institute on AgingNational Institutes of HealthParkinson's UK
KeywordsC9orf72Frontotemporal dementiaAmyotrophic lateral sclerosisStructural variationGeneticsBiologyDementiaDementia with Lewy bodiesLocus (genetics)Lewy bodyComputational biologyDiseaseGenomeNeuroscienceGeneMedicinePathology

Abstract

fetched live from OpenAlex

We characterized the role of structural variants, a largely unexplored type of genetic variation, in two non-Alzheimer’s dementias, namely Lewy body dementia (LBD) and frontotemporal dementia (FTD)/amyotrophic lateral sclerosis (ALS). To do this, we applied an advanced structural variant calling pipeline (GATK-SV) to short-read whole-genome sequence data from 5,213 European-ancestry cases and 4,132 controls. We discovered, replicated, and validated a deletion in TPCN1 as a novel risk locus for LBD and detected the known structural variants at the C9orf72 and MAPT loci as associated with FTD/ALS. We also identified rare pathogenic structural variants in both LBD and FTD/ALS. Finally, we assembled a catalog of structural variants that can be mined for new insights into the pathogenesis of these understudied forms of dementia.

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)0.002
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 score0.003
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0020.001
Science and technology studies0.0000.000
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 head0.037
GPT teacher head0.301
Teacher spread0.264 · 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".

Quick stats

Citations32
Published2023
Admission routes1
Has abstractyes

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