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Record W4401833050 · doi:10.1038/s41531-025-01245-z

Genome-wide association study of copy number variations in Parkinson’s disease

2024· preprint· en· W4401833050 on OpenAlexafffund
Zied Landoulsi, Ashwin Ashok Kumar Sreelatha, Nicole Kuznetsov, Dheeraj Reddy Bobbili, Ludovica Montanucci, Costin Leu, Lisa‐Marie Niestroj, Emadeldin Hassanin, Cloé Domenighetti, Pierre‐Emmanuel Sugier, Milena Radivojkov‐Blagojevic, Peter Lichtner, Berta Portugal, Connor Edsall, Jens Krüger, Dena Hernández, 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, Akiyoshi Nakayama, Nobutaka Hattori, Kenya Nishioka, Sun Ju Chung, Yun Joong Kim, Lukas Pavelka, Pierre Kolber, Andrew Singleton, Dan Vitale, Mathias Toft, Lasse Pihlstrøm, Leonor Correia Guedes, Joaquim J. Ferreira, Jonathan Carr, E. Tolosa, Mario Ezquerra, Pau Pástor, Karin Wirdefeldt, Nancy L. Pedersen, Caroline Ran, Andrea Carmine Belin, Andreas Puschmann, Carl E Clarke, Karen Morrison, Dimitri Krainc, Matthew J. Farrer, Dennis Lal, Thomas Gasser, Rejko Krüger, Manu Sharma, Patrick May

Bibliographic record

Venuenpj Parkinson s Disease · 2024
Typepreprint
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomic variations and chromosomal abnormalities
Canadian institutionsOntario Brain InstituteToronto Western HospitalOccupational Cancer Research CentreUniversity of Toronto
FundersMedical Research CouncilMinisterio de Ciencia e InnovaciónMultiple Sclerosis Society of Western AustraliaUniversiteit StellenboschVetenskapsrådetMinistero della SaluteAssociation France ParkinsonAgence Nationale de la RechercheSouth African Medical Research CouncilDeutsche ForschungsgemeinschaftEesti TeadusagentuurNational Research FoundationParkinson's UKDepartment of Science and Technology, Ministry of Science and Technology, IndiaMultiple System Atrophy CoalitionConsortium canadien en neurodégénérescence associée au vieillissementFaculty of Medicine and Health, University of SydneyKarolinska InstitutetEU Joint Programme – Neurodegenerative Disease ResearchMichael J. Fox Foundation for Parkinson's ResearchNational Institutes of HealthU.S. Department of Health and Human Services
KeywordsCopy-number variationGeneticsDiseaseGenome-wide association studyParkinson's diseaseBiologyGenetic associationGenomePathogenesisComputational biologyGeneSingle-nucleotide polymorphismMedicineGenotypePathology

Abstract

fetched live from OpenAlex

Abstract Objective To investigate the impact of copy number variations (CNVs) on Parkinson’s disease (PD) pathogenesis using genome-wide data and explore their role in sporadic PD. Methods We analyzed CNV data from 11,035 PD patients (including 2,731 early-onset PD (EOPD)) and 8,901 controls from the COURAGE-PD consortium using a sliding window CNV-GWAS and genome-wide burden analysis. The independent dataset from the Global Parkinson Genetics Program (GP2) consisted of 23,089 cases and 18,824 controls were used to validate our initial findings. Results The exploratory dataset identifies multiple CNV regions associated with PD risk. The nominated CNV loci were not confirmed in an independent dataset, except that only a deletion in the PRKN gene, a well-established EOPD locus, remained genome-wide significant and robustly supported. CNV burden analysis showed a higher prevalence of CNVs in PD-related genes in patients compared to controls (OR=1.56 [1.18-2.09], p=0.0013), with PRKN showing the highest burden (OR=1.47 [1.10-1.98], p=0.026). Patients with CNVs in PRKN had an earlier disease onset. Burden analysis with controls and EOPD patients showed similar results. Interpretation The largest CNV-based GWAS on PD highlights both the promise and pitfalls of array-based CNV detection in PD and underscores the relevance of whole-genome sequencing approaches in resolving the role of CNV in PD. The array-based findings are prone towards false positive findings that might arise either from platform limitations and/or cohort biases. Future studies require improved genotyping resolution and rigorous cross-cohort validation to reliably assess CNV contributions to PD risk.

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)0.005
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 score0.003
Threshold uncertainty score0.007

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.005
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0020.002
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.001
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 head0.011
GPT teacher head0.254
Teacher spread0.243 · 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
Published2024
Admission routes2
Has abstractyes

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