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Enregistrement W1619053513 · doi:10.1001/jamaneurol.2015.1700

Association of Long Runs of Homozygosity With Alzheimer Disease Among African American Individuals

2015· article· en· W1619053513 sur OpenAlex
Mahdi Ghani, Christiane Reitz, Rong Cheng, Badri N. Vardarajan, Gyungah Jun, Christine Sato, Adam C. Naj, Ruchita Rajbhandary, Li San Wang, Otto Valladares, Chiao‐Feng Lin, Eric B. Larson, Neill R. Graff‐Radford, Denis A. Evans, Philip L. De Jager, Paul K. Crane, Joseph D. Buxbaum, Jill R. Murrell, Towfique Raj, Nilüfer Ertekin‐Taner, Mark Logue, Clinton T. Baldwin, Robert C. Green, Lisa L. Barnes, Laura B. Cantwell, M. Daniele Fallin, Rodney C.P. Go, Patrick Griffith, Thomas O. Obisesan, Jennifer J. Manly, Kathryn L. Lunetta, M. Ilyas Kamboh, Oscar L. López, David A. Bennett, Hugh C. Hendrie, Kathleen Hall, Alison Goate, Goldie S. Byrd, Walter A. Kukull, Tatiana M. Foroud, Jonathan L. Haines, Lindsay A. Farrer, Margaret A. Pericak‐Vance, Joseph H. Lee, Gerard D. Schellenberg, Peter St George‐Hyslop, Richard Mayeux, Ekaterina Rogaeva

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Notice bibliographique

RevueJAMA Neurology · 2015
Typearticle
Langueen
DomaineMedicine
ThématiqueAlzheimer's disease research and treatments
Établissements canadiensOccupational Cancer Research CentreUniversity of Toronto
Organismes subventionnairesNational Center for Research ResourcesNational Cancer InstituteNational Human Genome Research InstituteNational Institute on AgingGeriatric Research Education and Clinical CenterNational Center for Advancing Translational SciencesMedical Research CouncilDepartment of Pathology, Johns Hopkins UniversitySchool of Medicine, Indiana UniversityUniversity of California, IrvineNational Institute of Mental HealthFeinberg School of MedicinePerelman School of Medicine, University of PennsylvaniaUniversity of California, DavisCanadian Institutes of Health ResearchUniversity of California, Los AngelesNational Institutes of HealthUniversity of California, San FranciscoAlzheimer SocietyUniversity of South FloridaNational Alzheimer's Coordinating CenterNational Institute on Minority Health and Health DisparitiesUSF Health Byrd Alzheimer's InstituteJohns Hopkins UniversityUniversity of WashingtonUniversity of MiamiYork UniversityNorthwestern UniversityCleveland ClinicEmory UniversityUniversity of PennsylvaniaVanderbilt UniversityChildren's Hospital of PhiladelphiaRush UniversityUniversity of California, San DiegoOffice of Research and DevelopmentU.S. Department of Veterans AffairsHope Center for Neurological DisordersWellcome TrustUniversity of Southern CaliforniaMassachusetts General HospitalGlaxoSmithKlineNational Institute of Neurological Disorders and StrokeUniversity of PittsburghUniversity of Colorado School of Medicine, Anschutz Medical CampusAlzheimer's Association
Mots-clésRuns of HomozygosityGeneticsSingle-nucleotide polymorphismGenotypeSNPBiologyGenome-wide association studyInbreedingPopulationAlleleGenetic associationDiseaseAllele frequencyLocus (genetics)MedicineGeneInternal medicine

Résumé

récupéré en direct d'OpenAlex

IMPORTANCE: Mutations in known causal Alzheimer disease (AD) genes account for only 1% to 3% of patients and almost all are dominantly inherited. Recessive inheritance of complex phenotypes can be linked to long (>1-megabase [Mb]) runs of homozygosity (ROHs) detectable by single-nucleotide polymorphism (SNP) arrays. OBJECTIVE: To evaluate the association between ROHs and AD in an African American population known to have a risk for AD up to 3 times higher than white individuals. DESIGN, SETTING, AND PARTICIPANTS: Case-control study of a large African American data set previously genotyped on different genome-wide SNP arrays conducted from December 2013 to January 2015. Global and locus-based ROH measurements were analyzed using raw or imputed genotype data. We studied the raw genotypes from 2 case-control subsets grouped based on SNP array: Alzheimer's Disease Genetics Consortium data set (871 cases and 1620 control individuals) and Chicago Health and Aging Project-Indianapolis Ibadan Dementia Study data set (279 cases and 1367 control individuals). We then examined the entire data set using imputed genotypes from 1917 cases and 3858 control individuals. MAIN OUTCOMES AND MEASURES: The ROHs larger than 1 Mb, 2 Mb, or 3 Mb were investigated separately for global burden evaluation, consensus regions, and gene-based analyses. RESULTS: The African American cohort had a low degree of inbreeding (F ~ 0.006). In the Alzheimer's Disease Genetics Consortium data set, we detected a significantly higher proportion of cases with ROHs greater than 2 Mb (P = .004) or greater than 3 Mb (P = .02), as well as a significant 114-kilobase consensus region on chr4q31.3 (empirical P value 2 = .04; ROHs >2 Mb). In the Chicago Health and Aging Project-Indianapolis Ibadan Dementia Study data set, we identified a significant 202-kilobase consensus region on Chr15q24.1 (empirical P value 2 = .02; ROHs >1 Mb) and a cluster of 13 significant genes on Chr3p21.31 (empirical P value 2 = .03; ROHs >3 Mb). A total of 43 of 49 nominally significant genes common for both data sets also mapped to Chr3p21.31. Analyses of imputed SNP data from the entire data set confirmed the association of AD with global ROH measurements (12.38 ROHs >1 Mb in cases vs 12.11 in controls; 2.986 Mb average size of ROHs >2 Mb in cases vs 2.889 Mb in controls; and 22% of cases with ROHs >3 Mb vs 19% of controls) and a gene-cluster on Chr3p21.31 (empirical P value 2 = .006-.04; ROHs >3 Mb). Also, we detected a significant association between AD and CLDN17 (empirical P value 2 = .01; ROHs >1 Mb), encoding a protein from the Claudin family, members of which were previously suggested as AD biomarkers. CONCLUSIONS AND RELEVANCE: To our knowledge, we discovered the first evidence of increased burden of ROHs among patients with AD from an outbred African American population, which could reflect either the cumulative effect of multiple ROHs to AD or the contribution of specific loci harboring recessive mutations and risk haplotypes in a subset of patients. Sequencing is required to uncover AD variants in these individuals.

Récupéré en direct depuis OpenAlex et désinversé. Les résumés ne sont pas conservés dans cette base de données : les index inversés représentent 8,6 Go des 9,3 Go de texte de la base, et le serveur dispose de 13 Go libres.

Prédiction distillée sur la base complète

Imitation des enseignants

Ni prévalence calibrée, ni vérité terrain. Validation humaine à venir. Apprise à partir de 10 348 étiquettes directes de Codex et de 10 348 étiquettes directes de Gemma. Le mode candidate est l'union des têtes enseignantes seuillées; le consensus est leur intersection. Ces sorties portent le statut machine_predicted_unvalidated et ne sont ni des étiquettes humaines ni des étiquettes directes de modèles de pointe.

score de la tête « metaresearch » (Codex)0,000
score de la tête « metaresearch » (Gemma)0,000
Version: codex-gemma-dda1882f352aStatut de validation: machine_predicted_unvalidated
Catégories candidatesaucune
Catégories consensuellesaucune
DomaineSignal candidat: aucune · Signal consensuel: aucune
Devis d'étudeSignal candidat: Observationnel · Signal consensuel: Observationnel
GenreSignal candidat: Empirique · Signal consensuel: Empirique
Score de désaccord entre enseignants0,005
Score d'incertitude au seuil0,444

Scores Codex et Gemma par catégorie

CatégorieCodexGemma
Métarecherche0,0000,000
Méta-épidémiologie (sens strict)0,0000,000
Méta-épidémiologie (sens large)0,0000,000
Bibliométrie0,0000,000
Études des sciences et des technologies0,0000,000
Communication savante0,0000,000
Science ouverte0,0000,000
Intégrité de la recherche0,0000,000
Charge utile insuffisante (le modèle a refusé de juger)0,0000,000

Scores machine (provisoires)

Les deux têtes enseignantes du modèle étudiant, lues sur ce travail. Un score ordonne la base pour la relecture; il n'affirme jamais une catégorie, et le statut de validation accompagne chaque rangée tel quel.

Scores de référence d'un modèle non mature (critères de maturité non atteints, 7 itérations). Un score ordonne; il n'affirme jamais une catégorie.

Tête enseignante Opus0,025
Tête enseignante GPT0,292
Écart entre enseignants0,267 · la distance entre les deux têtes enseignantes sur ce seul travail
Statut de validationscore_only:v0-immature-baseline · tel quel depuis la passe de notation : score_only signifie que le nombre peut ordonner les travaux, et qu'aucune étiquette de catégorie n'en découle