MétaCan
Menu
← Back to cohort
Record W3175285480 · doi:10.1101/2021.06.30.450599

Genome-wide association study and functional validation implicates JADE1 in tauopathy

2021· preprint· en· W3175285480 on OpenAlexaff
Kurt Farrell, SoongHo Kim, Natalia Han, Megan A. Iida, Elias Gonzalez, Marcos Otero‐García, Jamie M. Walker, Tim Richardson, Alan E. Renton, Shea J. Andrews, Brian Fulton‐Howard, Jack Humphrey, Ricardo A. Vialle, Kathryn R. Bowles, Kristen Whitney, Diana K. Dangoor, Edoardo Marcora, Marco M. Hefti, Alicia Casella, Cheick A. Sissoko, Manav Kapoor, Gloriia Novikova, Evan Udine, Garrett Wong, Weijing Tang, Tushar Bhangale, Julie Hunkapiller, Gai Ayalon, Rob Graham, Jonathan D. Cherry, Etty Cortés, Valeriy Borukov, Ann C. McKee, Thor D. Stein, Jean‐Paul Vonsattel, Marla Gearing, Jonathan D. Glass, Juan C. Troncoso, Matthew P. Frosch, Bradley T. Hyman, Dennis W. Dickson, Melissa E. Murray, Johannes Attems, Margaret E. Flanagan, Qinwen Mao, M-Marsel Mesulam, Sandra Weıntraub, Randy Woltjer, Thao Pham, Julia Kofler, Julie A. Schneider, Lei Yu, Dushyant P. Purohit, Vahram Haroutunian, Patrick R. Hof, Sam Gandy, Mary Sano, Thomas G. Beach, Wayne W. Poon, Claudia H. Kawas, María M. Corrada, Robert A. Rissman, Jeff Metcalf, Sara Shuldberg, Bahar Salehi, Peter T. Nelson, John Q. Trojanowski, Edward B. Lee, David A. Wolk, Corey T. McMillan, Dirk Keene, Thomas J. Montine, Gábor G. Kovács, Mirjam I. Lutz, Peter Fischer, Richard J. Perrin, Nigel J. Cairns, Erin Franklin, Herbert T. Cohen, Maria Inmaculada Cobos Sillero, Bess Frost, Towfique Raj, Alison Goate, Charles L. White, John F. Crary

Bibliographic record

VenuebioRxiv (Cold Spring Harbor Laboratory) · 2021
Typepreprint
Languageen
FieldMedicine
TopicAlzheimer's disease research and treatments
Canadian institutionsOntario Brain InstituteUniversity of TorontoUniversity Health Network
FundersMcDonnell Center for Systems NeuroscienceCharles F. and Joanne Knight Alzheimer Disease Research Center, Washington University in St. LouisUniversity of California, San DiegoGenentechUniversity of PittsburghUniversity of Texas Southwestern Medical CenterNancy and Buster Alvord EndowmentRossy FoundationNorthwestern UniversityArizona Department of Health ServicesMichael J. Fox Foundation for Parkinson's ResearchOregon Health and Science UniversityEmory UniversityUniversity of Pennsylvania
KeywordsTauopathyProgressive supranuclear palsyBiologyNeurofibrillary tangleTau proteinGeneticsAlzheimer's diseaseNeurosciencePathologyNeurodegenerationAtrophyMedicineDiseaseSenile plaques

Abstract

fetched live from OpenAlex

Abstract Primary age-related tauopathy (PART) is a neurodegenerative tauopathy with features distinct from but also overlapping with Alzheimer disease (AD). While both exhibit Alzheimer-type temporal lobe neurofibrillary degeneration alongside amnestic cognitive impairment, PART develops independently of amyloid-β (Aβ) deposition in plaques. The pathogenesis of PART is unknown, but evidence suggests it is associated with genes that promote tau pathology as well as others that protect from Aβ toxicity. Here, we performed a genetic association study in an autopsy cohort of individuals with PART ( n =647) using Braak neurofibrillary tangle stage as a quantitative trait adjusting for sex, age, genotyping platform, and principal components. We found significant associations with some candidate loci associated with AD and progressive supranuclear palsy, a primary tauopathy ( SLC24A4, MS4A6A, HS3ST1, MAPT and EIF2AK3 ). Genome-wide association analysis revealed a novel significant association with a single nucleotide polymorphism on chromosome 4 (rs56405341) in a locus containing three genes, including JADE1 which was significantly upregulated in tangle-bearing neurons by single-soma RNA-seq. Immunohistochemical studies using antisera targeting JADE1 protein revealed localization within tau aggregates in autopsy brain from tauopathies containing isoforms with four microtubule-binding domain repeats (4R) and mixed 3R/4R, but not with 3R exclusively. Co-immunoprecipitation revealed a direct and specific binding of JADE1 protein to tau containing four (4R) and no N-terminal inserts (0N4R) in post-mortem human PART brain tissue. Finally, knockdown of the Drosophila JADE1 homolog rhinoceros (rno) enhanced tau-induced toxicity and apoptosis in vivo in a humanized 0N4R mutant tau knock-in model as quantified by rough eye phenotype and terminal deoxynucleotidyl transferase dUTP nick end-labeling (TUNEL) in the fly brain. Together, these findings indicate that PART has a genetic architecture that partially overlaps with AD and other tauopathies and suggests a novel role for JADE1 as a mediator of neurofibrillary degeneration.

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 score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0010.001
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 head0.023
GPT teacher head0.262
Teacher spread0.239 · 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

Citations2
Published2021
Admission routes1
Has abstractyes

Explore more

Same venuebioRxiv (Cold Spring Harbor Laboratory)→Same topicAlzheimer's disease research and treatments→French-language works237,207→