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Record W4366162959 · doi:10.1038/s41591-023-02268-w

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

2023· article· en· W4366162959 on OpenAlexafffund
Marie‐Gabrielle Duperron, Maria J. Knol, Quentin Le Grand, Tavia E. Evans, Aniket Mishra, Ami Tsuchida, 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, Nicola J. Armstrong, Constance Bordes, Mark E. Bastin, Alexa Beiser, Henry Brodaty, Robin Bülow, Caty Carrera, Christopher Chen, Ching‐Yu Cheng, Ian J. Deary, Piyush Gampawar, Jayandra J. Himali, Jiyang Jiang, Takahisa Kawaguchi, Shuo Li, Mélissa Macalli, Pascale Marquis, Zoë Morris, Susana Muñoz Maniega, Susumu Miyamoto, Masakazu Okawa, Matthew Paradise, Pedram Parva, Tatjana Rundek, Muralidharan Sargurupremraj, Sabrina Schilling, Kazuya Setoh, Omar Soukarieh, Yasuharu Tabara, Alexander Teumer, Anbupalam Thalamuthu, Julian N. Trollor, Maria C. Valdés Hernández, Meike W. Vernooij, Uwe Völker, Katharina Wittfeld, Tien Yin Wong, Margaret J. Wright, Junyi Zhang, Wanting Zhao, Yi‐Cheng Zhu, Helena Schmidt, Perminder S. Sachdev, Wei Wen, Kazumichi Yoshida, Anne Joutel, Claudia L. Satizábal, Ralph L. Sacco, Guillaume Bourque, Mark Lathrop, Tomáš Paus, Israel Fernández‐Cadenas, Qiong Yang, Bernard Mazoyer, Philippe Boutinaud, Yukinori Okada, Hans J. Grabe, Karen A. Mather, Reinhold Schmidt, Marc Joliot, M. Arfan Ikram, Fumihiko Matsuda, Christophe Tzourio, Joanna M. Wardlaw, Sudha Seshadri, Hieab H.H. Adams, Stéphanie Debette

Bibliographic record

VenueNature Medicine · 2023
Typearticle
Languageen
FieldNeuroscience
TopicCerebrospinal fluid and hydrocephalus
Canadian institutionsUniversité de MontréalCentre Hospitalier Universitaire Sainte-JustineUniversity of TorontoSickKids FoundationHospital for Sick ChildrenMcGill University
FundersNational Institute of Neurological Disorders and StrokeNational Institute for Dementia ResearchNational Institute on AgingEuropean Regional Development FundAlzheimer's SocietyCanadian Institutes of Health ResearchAgence Nationale de la RechercheUniversität GreifswaldInstituto de Salud Carlos IIIUK Dementia Research InstituteCentre for Cognitive Ageing and Cognitive EpidemiologySiemens HealthineersMcKnight FoundationServierMedical Research CouncilNational Health and Medical Research CouncilNeuraxpharmAustrian Science FundMedizinische Universität GrazMinistry of Cultural AffairsUniversité de LilleOesterreichische NationalbankDirectorate for Biological SciencesMutuelle Générale de l'Education NationaleNational Heart, Lung, and Blood InstituteBiotechnology and Biological Sciences Research CouncilNational Medical Research CouncilFondation Bettencourt SchuellerNational University Health SystemSanofiFondation de FranceFondation pour la Recherche MédicaleZonMwNederlandse Organisatie voor Wetenschappelijk OnderzoekBundesministerium für Bildung und ForschungMinistry of Education, Culture, Sports, Science and TechnologyFondation LeducqBundesministerium für Wissenschaft, Forschung und WirtschaftJapan Society for the Promotion of ScienceUniversity of EdinburghCilagUniversité de BordeauxGenome CanadaCompute CanadaAge UKFresenius Medical Care North AmericaDevelopment of Innovative Strategies for a Transdisciplinary approach to ALZheimer's diseaseScottish Funding CouncilErasmus Medisch CentrumNeuroscience Research AustraliaMrs Gladys Row Fogo Charitable TrustKarl-Franzens-Universität GrazJapan Agency for Medical Research and DevelopmentMcGill UniversityEuropean CommissionInstitut National de la Santé et de la Recherche MédicaleEU Joint Programme – Neurodegenerative Disease Research
KeywordsWhite matterPerivascular spaceMendelian randomizationLeukodystrophyPopulationGenome-wide association studyStroke (engine)BiologyPathologyDiseaseCADASILMagnetic resonance imagingMedicineNeuroscienceDementiaGeneticsGeneSingle-nucleotide polymorphism

Abstract

fetched live from OpenAlex

Perivascular space (PVS) burden is an emerging, poorly understood, magnetic resonance imaging marker of cerebral small vessel disease, a leading cause of stroke and dementia. Genome-wide association studies in up to 40,095 participants (18 population-based cohorts, 66.3 ± 8.6 yr, 96.9% European ancestry) revealed 24 genome-wide significant PVS risk loci, mainly in the white matter. These were associated with white matter PVS already in young adults (N = 1,748; 22.1 ± 2.3 yr) and were enriched in early-onset leukodystrophy genes and genes expressed in fetal brain endothelial cells, suggesting early-life mechanisms. In total, 53% of white matter PVS risk loci showed nominally significant associations (27% after multiple-testing correction) in a Japanese population-based cohort (N = 2,862; 68.3 ± 5.3 yr). Mendelian randomization supported causal associations of high blood pressure with basal ganglia and hippocampal PVS, and of basal ganglia PVS and hippocampal PVS with stroke, accounting for blood pressure. Our findings provide insight into the biology of PVS and cerebral small vessel disease, pointing to pathways involving extracellular matrix, membrane transport and developmental processes, and the potential for genetically informed prioritization of drug targets.

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)0.000
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 score0.004

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.000
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 head0.018
GPT teacher head0.259
Teacher spread0.241 · 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".

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Citations123
Published2023
Admission routes2
Has abstractyes

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