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Record W4281609742 · doi:10.1212/wnl.0000000000200699

Genome-wide Association and Meta-analysis of Age at Onset in Parkinson Disease

2022· review· en· W4281609742 on OpenAlexfundno aff
Sandeep Grover, Ashwin Ashok Kumar Sreelatha, Lasse Pihlstrøm, Cloé Domenighetti, Claudia Schulte, Pierre‐Emmanuel Sugier, Milena Radivojkov‐Blagojevic, Peter Lichtner, Océane Mohamed, Berta Portugal, Zied Landoulsi, Patrick May, Dheeraj Reddy Bobbili, Connor Edsall, Felix Bartusch, Maximilian Hanussek, Jens Krüger, Dena G. Hernandez, Cornelis Blauwendraat, George D. Mellick, Alexander Zimprich, Walter Pirker, Manuela Tan, Ekaterina Rogaeva, Anthony E. Lang, Sulev Kõks, Pille Taba, Suzanne Lesage, Alexis Brice, Jean‐Christophe Corvol, Marie‐Christine Chartier‐Harlin, Eugénie Mutez, Kathrin Brockmann, Angela Deutschländer, G. Hadjigeorgiou, Efthimos Dardiotis, Leonidas Stefanis, Athina Maria Simitsi, Enza Maria Valente, Simona Petrucci, Letizia Straniero, Anna Zecchinelli, Gianni Pezzoli, Laura Brighina, Carlo Ferrarese, Grazia Annesi, Andrea Quattrone, Monica Gagliardi, Lena F. Burbulla, Hirotaka Matsuo, Yusuke Kawamura, Nobutaka Hattori, Kenya Nishioka, Sun Ju Chung, Yun Joong Kim, Lukas Pavelka, Bart P.C. van de Warrenburg, Bastiaan R. Bloem, Andrew Singleton, Jan Aasly, Mathias Toft, Leonor Correia Guedes, Joaquim J. Ferreira, Soraya Bardien, Jonathan Carr, Eduardo Tolosa, Mario Ezquerra, Pau Pástor, Mónica Díez-Fairén, Karin Wirdefeldt, Nancy L. Pedersen, Caroline Ran, Andrea Carmine Belin, Andreas Puschmann, Clara Hellberg, Carl E Clarke, Karen Morrison, Dimitri Krainc, Matthew J. Farrer, Rejko Krüger, Alexis Elbaz, Thomas Gasser, Manu Sharma

Bibliographic record

VenueNeurology · 2022
Typereview
Languageen
FieldMedicine
TopicParkinson's Disease Mechanisms and Treatments
Canadian institutionsnot available
FundersSorbonne UniversitéDeutsches Zentrum für Neurodegenerative ErkrankungenUniversity of TorontoAssistance Publique - Hôpitaux de ParisFeinberg School of MedicineTartu ÜlikoolNational Defense Medical CollegeMurdoch UniversityNorthwestern University
KeywordsGenome-wide association studyCourageGenetic associationMeta-analysisParkinson's diseaseHeritabilityDiseaseGeneticsMedicineBiologyInternal medicineSingle-nucleotide polymorphismGenotypeGene

Abstract

fetched live from OpenAlex

<h3>Background and Objectives</h3> Considerable heterogeneity exists in the literature concerning genetic determinants of the age at onset (AAO) of Parkinson disease (PD), which could be attributed to a lack of well-powered replication cohorts. The previous largest genome-wide association studies (GWAS) identified <i>SNCA</i> and <i>TMEM175</i> loci on chromosome (Chr) 4 with a significant influence on the AAO of PD; these have not been independently replicated. This study aims to conduct a meta-analysis of GWAS of PD AAO and validate previously observed findings in worldwide populations. <h3>Methods</h3> A meta-analysis was performed on PD AAO GWAS of 30 populations of predominantly European ancestry from the Comprehensive Unbiased Risk Factor Assessment for Genetics and Environment in Parkinson9s Disease (COURAGE-PD) Consortium. This was followed by combining our study with the largest publicly available European ancestry dataset compiled by the International Parkinson Disease Genomics Consortium (IPDGC). <h3>Results</h3> The COURAGE-PD Consortium included a cohort of 8,535 patients with PD (91.9%: Europeans and 9.1%: East Asians). The average AAO in the COURAGE-PD dataset was 58.9 years (SD = 11.6), with an underrepresentation of females (40.2%). The heritability estimate for AAO in COURAGE-PD was 0.083 (SE = 0.057). None of the loci reached genome-wide significance (<i>p</i> &lt; 5 × 10<sup>−8</sup>). Nevertheless, the COURAGE-PD dataset confirmed the role of the previously published <i>TMEM175</i> variant as a genetic determinant of the AAO of PD with Bonferroni-corrected nominal levels of significance (<i>p</i> &lt; 0.025): (rs34311866: β(SE)<sub>COURAGE</sub> = 0.477(0.203), <i>p</i><sub>COURAGE</sub> = 0.0185). The subsequent meta-analysis of COURAGE-PD and IPDGC datasets (N<sub>total</sub> = 25,950) led to the identification of 2 genome-wide significant association signals on Chr 4, including the previously reported <i>SNCA</i> locus (rs983361: β(SE)<sub>COURAGE+IPDGC</sub> = 0.720(0.122), <i>p</i><sub>COURAGE+IPDGC</sub> = 3.13 × 10<sup>−9</sup>) and a novel <i>BST1</i> locus (rs4698412: β(SE)<sub>COURAGE+IPDGC</sub> = −0.526(0.096), <i>p</i><sub>COURAGE+IPDGC</sub> = 4.41 × 10<sup>−8</sup>). <h3>Discussion</h3> Our study further refines the genetic architecture of Chr 4 underlying the AAO of the PD phenotype through the identification of <i>BST1</i> as a novel AAO PD locus. These findings open a new direction for the development of treatments to delay the onset of PD.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow), Insufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Meta-analysis · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.551
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0040.002
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
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 head0.075
GPT teacher head0.324
Teacher spread0.250 · 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 teacher head, not a consensus.

Study designMeta-analysis
Domainnot available
GenreReview

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

Citations71
Published2022
Admission routes1
Has abstractyes

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