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Record W2895909123 · doi:10.1101/424010

Parkinson disease age of onset GWAS: defining heritability, genetic loci and a-synuclein mechanisms

2018· preprint· en· W2895909123 on OpenAlexafffund
Cornelis Blauwendraat, Karl Heilbron, Costanza L. Vallerga, Sara Bandrés‐Ciga, Rainer von Coelln, Lasse Pihlstrøm, Javier Simón‐Sánchez, Claudia Schulte, Manu Sharma, Lynne Krohn, Ari Siitonen, Hirotaka Iwaki, Hampton L. Leonard, Alastair J. Noyce, Manuela Tan, J. Raphael Gibbs, Dena G. Hernandez, Sonja W. Scholz, Joseph Jankovic, Lisa M. Shulman, Suzanne Lesage, Jean‐Christophe Corvol, Alexis Brice, Jacobus J. van Hilten, Johan Marinus, Pentti J. Tienari, Kari Majamaa, Mathias Toft, Donald G. Grosset, Thomas Gasser, Peter Heutink, Joshua Shulman, John Hardy, Huw R. Morris, David A. Hinds, Jacob Gratten, Peter M. Visscher, Ziv Gan‐Or, Mike A. Nalls, Andrew Singleton

Bibliographic record

VenuebioRxiv (Cold Spring Harbor Laboratory) · 2018
Typepreprint
Languageen
FieldMedicine
TopicParkinson's Disease Mechanisms and Treatments
Canadian institutionsCentre for Movement DisordersMcGill UniversityMontreal Neurological Institute and Hospital
FundersMedical Research CouncilNational Institute of Neurological Disorders and StrokeBundesministerium für Bildung und ForschungMultiple System Atrophy CoalitionNational Institute on AgingConsortium canadien en neurodégénérescence associée au vieillissementWellcome TrustDeutsche ForschungsgemeinschaftHelsingin ja Uudenmaan SairaanhoitopiiriHelsingin YliopistoNational Institute of Environmental Health SciencesEU Joint Programme – Neurodegenerative Disease ResearchMichael J. Fox Foundation for Parkinson's ResearchAmerican Parkinson Disease AssociationItä-Suomen YliopistoInstitute of GeneticsNational Institutes of HealthU.S. Department of Health and Human ServicesMcGill UniversityU.S. Department of Defense
KeywordsGenome-wide association studyHeritabilityGenetic associationMissing heritability problemLocus (genetics)GeneticsAge of onsetBiologyDiseaseBonferroni correctionGenetic variationGenotypeMedicineSingle-nucleotide polymorphismInternal medicineGene

Abstract

fetched live from OpenAlex

Abstract Increasing evidence supports an extensive and complex genetic contribution to Parkinson’s disease (PD). Previous genome-wide association studies (GWAS) have shed light on the genetic basis of risk for this disease. However, the genetic determinants of PD age of onset are largely unknown. Here we performed an age of onset GWAS based on 28,568 PD cases. We estimated that the heritability of PD age of onset due to common genetic variation was ~0.11, lower than the overall heritability of risk for PD (~0.27) likely in part because of the subjective nature of this measure. We found two genome-wide significant association signals, one at SNCA and the other a protein-coding variant in TMEM175 , both of which are known PD risk loci and a Bonferroni corrected significant effect at other known PD risk loci, INPP5F/BAG3 , FAM47E/SCARB2 , and MCCC1. In addition, we identified that GBA coding variant carriers had an earlier age of onset compared to non-carriers. Notably, SNCA , TMEM175 , SCARB2 , BAG3 and GBA have all been shown to either directly influence alpha-synuclein aggregation or are implicated in alpha-synuclein aggregation pathways. Remarkably, other well-established PD risk loci such as GCH1, MAPT and RAB7L1 /NUCKS1 (PARK16) did not show a significant effect on age of onset of PD. While for some loci, this may be a measure of power, this is clearly not the case for the MAPT locus; thus genetic variability at this locus influences whether but not when an individual develops disease. We believe this is an important mechanistic and therapeutic distinction. Furthermore, these data support a model in which alpha-synuclein and lysosomal mechanisms impact not only PD risk but also age of disease onset and highlights that therapies that target alpha-synuclein aggregation are more likely to be disease-modifying than therapies targeting other pathways.

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)0.002
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 score0.011

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.004
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.002
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.001
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 head0.016
GPT teacher head0.234
Teacher spread0.218 · 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

Citations12
Published2018
Admission routes2
Has abstractyes

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