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Record W3003184985 · doi:10.1186/s40478-020-0879-z

Analysis of neurodegenerative disease-causing genes in dementia with Lewy bodies

2020· article· en· W3003184985 on OpenAlexafffund
Tatiana Orme, Dena G. Hernandez, Owen A. Ross, Célia Kun‐Rodrigues, Lee Darwent, Claire E. Shepherd, Laura Parkkinen, Olaf Ansorge, Lorraine N. Clark, Lawrence S. Honig, Karen Marder, Afina W. Lemstra, Ekaterina Rogaeva, Peter St George‐Hyslop, Elisabet Londos, Henrik Zetterberg, Kevin Morgan, Claire Troakes, Safa Al‐Sarraj, Tammaryn Lashley, Janice L. Holton, Yaroslau Compta, Vivianna M. Van Deerlin, John Q. Trojanowski, Geidy E. Serrano, Thomas G. Beach, Suzanne Lesage, Douglas Galasko, Eliezer Masliah, Isabel Santana, Pau Pástor, Pentti J. Tienari, Liisa Myllykangas, Minna Oinas, Tamás Révész, Andrew J. Lees, Bradley F. Boeve, Ronald C. Petersen, Tanis J. Ferman, Valentina Escott‐Price, Caroline Graff, Nigel J. Cairns, John C. Morris, Stuart Pickering‐Brown, David Mann, Glenda M. Halliday, David J. Stone, Dennis W. Dickson, John Hardy, Andrew Singleton, Rita Guerreiro, José Brás

Bibliographic record

VenueActa Neuropathologica Communications · 2020
Typearticle
Languageen
FieldNeuroscience
TopicNeurological diseases and metabolism
Canadian institutionsUniversity of Toronto
FundersNational Institute of Neurological Disorders and StrokeNIHR Oxford Biomedical Research CentreNational Center for Advancing Translational SciencesMedical Research CouncilArizona Biomedical Research CommissionUniversity of California, San DiegoUniversity of New South WalesFolkhälsanin TutkimussäätiöUniversity College London Hospitals NHS Foundation TrustNational Institutes of HealthRosetrees TrustParkinson's UKNational Institute on AgingNational Institute for Health and Care ResearchAgence Nationale de la RechercheAlzheimer SocietyAlzheimer's SocietyNeuroscience Research AustraliaMichael J. Fox Foundation for Parkinson's ResearchArizona Department of Health ServicesUniversity of PennsylvaniaLittle Family FoundationHelsingin ja Uudenmaan SairaanhoitopiiriParkinson's Disease FoundationConsortium canadien en neurodégénérescence associée au vieillissementSamfundet FolkhälsanMayo ClinicU.S. Department of Health and Human Services
KeywordsDementia with Lewy bodiesFrontotemporal dementiaDementiaExome sequencingMissense mutationDiseaseMedicineNeurologyGeneticsBiologyMutationGenePathologyPsychiatry

Abstract

fetched live from OpenAlex

Dementia with Lewy bodies (DLB) is a clinically heterogeneous disorder with a substantial burden on healthcare. Despite this, the genetic basis of the disorder is not well defined and its boundaries with other neurodegenerative diseases are unclear. Here, we performed whole exome sequencing of a cohort of 1118 Caucasian DLB patients, and focused on genes causative of monogenic neurodegenerative diseases. We analyzed variants in 60 genes implicated in DLB, Alzheimer's disease, Parkinson's disease, frontotemporal dementia, and atypical parkinsonian or dementia disorders, in order to determine their frequency in DLB. We focused on variants that have previously been reported as pathogenic, and also describe variants reported as pathogenic which remain of unknown clinical significance, as well as variants associated with strong risk. Rare missense variants of unknown significance were found in APP, CHCHD2, DCTN1, GRN, MAPT, NOTCH3, SQSTM1, TBK1 and TIA1. Additionally, we identified a pathogenic GRN p.Arg493* mutation, potentially adding to the diversity of phenotypes associated with this mutation. The rarity of previously reported pathogenic mutations in this cohort suggests that the genetic overlap of other neurodegenerative diseases with DLB is not substantial. Since it is now clear that genetics plays a role in DLB, these data suggest that other genetic loci play a role in this disease.

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)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.856
Threshold uncertainty score0.620

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.002
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.001
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 head0.113
GPT teacher head0.301
Teacher spread0.187 · 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

Citations42
Published2020
Admission routes2
Has abstractyes

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