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Record W4405700538 · doi:10.1002/alz.14444

Integrating rare pathogenic variant prioritization with gene‐based association analysis to identify novel genes and relevant multimodal traits for Alzheimer's disease

2024· article· en· W4405700538 on OpenAlexfundno aff
Jixin Cao, Cheng Zhang, Chun‐Yi Zac Lo, Qihao Guo, Jing Ding, Xiaohui Luo, Zeji Zhang, Feng Chen, Tian‐Lin Cheng, Jingqi Chen, Xing‐Ming Zhao

Bibliographic record

VenueAlzheimer s & Dementia · 2024
Typearticle
Languageen
FieldMedicine
TopicAlzheimer's disease research and treatments
Canadian institutionsnot available
FundersNational Institute of Biomedical Imaging and BioengineeringNational Institute on AgingNational Key Research and Development Program of ChinaNatural Science Foundation of ShanghaiCanadian Institutes of Health ResearchNational Institutes of HealthArizona Biomedical Research CommissionScience and Technology Commission of Shanghai MunicipalityNational Natural Science Foundation of ChinaNational Institute on Drug AbuseNational Key Laboratory of Human Factors EngineeringNational Institute of Neurological Disorders and StrokeNorthern California Institute for Research and EducationArizona Department of Health Services
KeywordsEndophenotypeCandidate geneBiologyDiseaseGenome-wide association studyGeneGenetic associationGeneticsComputational biologyBioinformaticsMedicineNeuroscienceGenotypeCognitionSingle-nucleotide polymorphismPathology

Abstract

fetched live from OpenAlex

INTRODUCTION: Increasing evidence has highlighted rare variants in Alzheimer's disease (AD). However, insufficient sample sizes, especially in underrepresented ethnic groups, hinder their investigation. Additionally, their impact on endophenotypes remains largely unexplored. METHODS: We prioritized rare likely-deleterious variants based on whole-genome sequencing data from a Chinese AD cohort (n = 988). Gene-based optimal sequence kernel association tests were conducted between AD cases and normal controls to identify AD-related genes. Network clustering, endophenotype association, and cellular experiments were conducted to evaluate their functional consequences. RESULTS: We identified 11 novel AD candidate genes, which captured AD-related pathways and enhanced AD risk prediction performance. Key genes (RABEP1, VIPR1, RPL3L, and CABIN1) were linked to cognitive decline and brain atrophy. Experiments showed RABEP1 p.R845W inducing endocytosis dysregulation and exacerbating toxic amyloid β accumulation, underscoring its therapeutic potential. DISCUSSION: Our findings highlighted the contributions of rare variants to AD and provided novel insights into AD therapeutics. HIGHLIGHTS: Identified 11 novel AD candidate genes in a Chinese AD cohort. Correlated candidate genes with AD-related cognitive and brain imaging traits. Indicated RABEP1 p.R845W as a critical AD contributor in the endocytic pathway.

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)0.003
metaresearch head score (Gemma)0.004
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 score0.004
Threshold uncertainty score0.016

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0030.004
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.002
Science and technology studies0.0010.000
Scholarly communication0.0010.000
Open science0.0010.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0030.000

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 head0.028
GPT teacher head0.325
Teacher spread0.297 · 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

Citations9
Published2024
Admission routes1
Has abstractyes

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