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Record W3201218752 · doi:10.1186/s40478-021-01250-2

Analysis of genes (TMEM106B, GRN, ABCC9, KCNMB2, and APOE) implicated in risk for LATE-NC and hippocampal sclerosis provides pathogenetic insights: a retrospective genetic association study

2021· article· en· W3201218752 on OpenAlexfundno aff
Adam Dugan, Peter T. Nelson, Yuriko Katsumata, Lincoln M. P. Shade, Kevin L. Boehme, Merilee Teylan, Matthew D. Cykowski, Shubhabrata Mukherjee, John S. K. Kauwe, Timothy J. Hohman, Julie A. Schneider, David W. Fardo

Bibliographic record

VenueActa Neuropathologica Communications · 2021
Typearticle
Languageen
FieldMedicine
TopicAlzheimer's disease research and treatments
Canadian institutionsnot available
FundersNational Center for Research ResourcesNational Institute of Neurological Disorders and StrokeNational Cancer InstituteNational Institute of Biomedical Imaging and BioengineeringNational Human Genome Research InstituteNational Institute on Drug AbuseNational Institute of Mental HealthNational Institute on AgingUniversity of California, San FranciscoStichting MS ResearchCanadian Institutes of Health ResearchPfizerUniversity of WashingtonUniversity of California, Los AngelesGenentechNational Institutes of HealthCommon FundH. Lundbeck A/SMedical Research CouncilServierNewcastle UniversityNorthern California Institute for Research and EducationUniversitat de BarcelonaF. Hoffmann-La RocheRush UniversityNational Center for Advancing Translational SciencesNovartis Pharmaceuticals CorporationUniversity of California, San DiegoJohns Hopkins UniversityNIH Office of the DirectorYork UniversityUniversity of MiamiNorthwestern UniversityBiogenBioClinicaEmory UniversityU.S. Department of Veterans AffairsUniversity of PennsylvaniaVanderbilt UniversityIXICOUniversity of PittsburghOffice of Research and DevelopmentUniversity of California, DavisFoundation for the National Institutes of HealthUniversity of Southern CaliforniaEisaiHoward Hughes Medical InstituteMassachusetts General HospitalAlzheimer's Research TrustU.S. Department of DefenseHersenstichtingEli Lilly and CompanyNational Heart, Lung, and Blood InstituteNorth Bristol NHS TrustBristol-Myers SquibbAlzheimer's AssociationWellcome TrustUniversity of California, IrvineMeso Scale Diagnostics
KeywordsHippocampal sclerosisGenome-wide association studyGenetic associationApolipoprotein EPhenotypeBiologySingle-nucleotide polymorphismDiseaseGenotypeGeneticsMedicinePathologyGeneNeuroscienceTemporal lobe

Abstract

fetched live from OpenAlex

Limbic-predominant age-related TDP-43 encephalopathy neuropathologic change (LATE-NC) is the most prevalent subtype of TDP-43 proteinopathy, affecting up to 1/3rd of aged persons. LATE-NC often co-occurs with hippocampal sclerosis (HS) pathology. It is currently unknown why some individuals with LATE-NC develop HS while others do not, but genetics may play a role. Previous studies found associations between LATE-NC phenotypes and specific genes: TMEM106B, GRN, ABCC9, KCNMB2, and APOE. Data from research participants with genomic and autopsy measures from the National Alzheimer's Coordinating Center (NACC; n = 631 subjects included) and the Religious Orders Study and Memory and the Rush Aging Project (ROSMAP; n = 780 included) were analyzed in the current study. Our goals were to reevaluate disease-associated genetic variants using newly collected data and to query whether the specific genotype/phenotype associations could provide new insights into disease-driving pathways. Research subjects included in prior LATE/HS genome-wide association studies (GWAS) were excluded. Single nucleotide variants (SNVs) within 10 kb of TMEM106B, GRN, ABCC9, KCNMB2, and APOE were tested for association with HS and LATE-NC, and separately for Alzheimer's pathologies, i.e. amyloid plaques and neurofibrillary tangles. Significantly associated SNVs were identified. When results were meta-analyzed, TMEM106B, GRN, and APOE had significant gene-based associations with both LATE and HS, whereas ABCC9 had significant associations with HS only. In a sensitivity analysis limited to LATE-NC + cases, ABCC9 variants were again associated with HS. By contrast, the associations of TMEM106B, GRN, and APOE with HS were attenuated when adjusting for TDP-43 proteinopathy, indicating that these genes may be associated primarily with TDP-43 proteinopathy. None of these genes except APOE appeared to be associated with Alzheimer's-type pathology. In summary, using data not included in prior studies of LATE or HS genomics, we replicated several previously reported gene-based associations and found novel evidence that specific risk alleles can differentially affect LATE-NC and HS.

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: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.009
Threshold uncertainty score0.651

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
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 head0.051
GPT teacher head0.319
Teacher spread0.267 · 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

Citations56
Published2021
Admission routes1
Has abstractyes

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