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Record W4312087534 · doi:10.1002/alz.064953

Genomics of perivascular space burden unravels early mechanisms of cerebral small vessel disease

2022· article· en· W4312087534 on OpenAlexaff
Marie‐Gabrielle Duperron, Maria J. Knol, Quentin Le Grand, Tavia E. Evans, Aniket Mishra, Gennady V. Roshchupkin, Takahiro Konuma, David‐Alexandre Trégouët, José R. Romero, Stefan Frenzel, Michelle Luciano, Edith Hofer, Mathieu Bourgey, Nicole Dueker, Pilar Delgado, Saima Hilal, Rick M. Tankard, Florian Dubost, Jean Shin, Yasaman Saba, Christopher Chen, Tatjana Rundek, Alexander Teumer, Ami Tsuchida, Helena Schmidt, Perminder S. Sachdev, Wei Wen, Anne Joutel, Claudia L. Satizábal, Ralph L. Sacco, Guillaume Bourque, Mark Lathrop, Tomáš Paus, Israel Fernández‐Cadenas, Bernard Mazoyer, Yukinori Okada, Hans J. Grabe, Karen A. Mather, Reinhold Schmidt, M. Arfan Ikram, Christophe Tzourio, Joanna M. Wardlaw, Sudha Seshadri, Hieab H.H. Adams, Stéphanie Debette

Bibliographic record

VenueAlzheimer s & Dementia · 2022
Typearticle
Languageen
FieldNeuroscience
TopicCerebrospinal fluid and hydrocephalus
Canadian institutionsUniversité de MontréalMcGill University and Génome Québec Innovation CentreUniversity of TorontoMcGill University
Fundersnot available
KeywordsMendelian randomizationGenome-wide association studyGenetic associationBiologyDiseaseMedicineBioinformaticsGeneticsInternal medicineGeneGenotypeSingle-nucleotide polymorphismGenetic variants

Abstract

fetched live from OpenAlex

Abstract Background Perivascular space (PVS) burden is an emerging MRI‐marker of cerebral small vessel disease (cSVD), a leading cause of stroke and dementia. Underlying mechanisms of PVS are unknown. PVS are thought to be related to the glymphatic system, involved in brain clearance of molecules such as amyloid beta. We aimed to decipher the genetic underpinnings of PVS burden. Method We conducted genome‐wide and whole‐exome association studies in N = 39,823 participants for white matter (WM) PVS, N = 40,000 for basal ganglia (BG) PVS and N = 40,095 for hippocampal (HIP) PVS (21 population‐based cohorts, 66.3±8.6 years). As PVS were rated with different scales across cohorts, we tested association of genetic variants with the top quartile of PVS burden distribution in each cohort followed by a sample‐size weighted meta‐analysis. We searched for shared genetic variation with related vascular and neurological phenotypes using linkage disequilibrium‐score regression, explored causality of associations with putative risk factors using Mendelian randomization and conducted extensive functional exploration of identified PVS loci using multiple bioinformatics approaches, including transcriptome‐wide association studies. Result We identified 24 genome‐wide significant PVS risk loci. These showed association with WM PVS already at age 20 in 1,748 young healthy adults, suggesting an important role of early‐life factors. PVS loci were enriched in genes causing early‐onset leukodystrophies and genes expressed in fetal brain endothelial cells. Mendelian randomization analyses supported causal associations of high blood pressure with BG and HIP PVS, and of BG PVS with stroke. Transcriptome‐wide association and colocalization analyses suggest causal implication of 11 genes, that could be prioritized for experimental follow‐up. Two‐thirds of PVS loci point to novel pathways, involving extracellular matrix, membrane transport, and developmental processes, with enrichment in targets of existing drugs for vascular cognitive, and infectious disorders. Conclusion In this first gene‐mapping study of PVS, one of the earliest MRI‐markers of cSVD, we describe 24 genome‐wide significant risk loci. Our findings provide completely novel insight into the biology of PVS across the adult lifespan and its contribution to cSVD pathophysiology, with potential for genetically informed prioritization of drug targets for prevention trials of cSVD, a major cause of stroke and dementia worldwide.

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 score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0020.000

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 head0.027
GPT teacher head0.236
Teacher spread0.209 · 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

Citations4
Published2022
Admission routes1
Has abstractyes

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