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Record W2764285200 · doi:10.1016/s1474-4422(17)30327-7

Identification of novel risk loci for restless legs syndrome in genome-wide association studies in individuals of European ancestry: a meta-analysis

2017· review· en· W2764285200 on OpenAlexaff
Barbara Schormair, Chen Zhao, Steven Bell, Erik Tilch, Aaro V. Salminen, Benno Pütz, Yves Dauvilliers, Ambra Stefani, Birgit Högl, Werner Poewe, David Kemlink, Karel Šonka, Cornelius G. Bachmann, Walter Paulus, Claudia Trenkwalder, Wolfgang H. Oertel, Magdolna Hornyak, Maris Teder‐Laving, Andres Metspalu, Georgios M. Hadjigeorgiou, Olli Polo, Ingo Fietze, Owen A. Ross, Zbigniew K. Wszołek, Adam S. Butterworth, Nicole Soranzo, Willem H. Ouwehand, David J. Roberts, John Danesh, Richard P. Allen, Christopher J. Earley, William G. Ondo, Lan Xiong, Jacques Montplaisir, Ziv Gan‐Or, Markus Perola, Pavel Vodička, Christian Dina, André Franke, Lukas Tittmann, Alexandre F.R. Stewart, Svati H. Shah, Christian Gieger, Annette Peters, Guy A. Rouleau, Klaus Berger, Konrad Oexle, Emanuele Di Angelantonio, David A. Hinds, Bertram Müller‐Myhsok, Juliane Winkelmann, Beverley Balkau, Pierre Ducimetière, Eveline Eschwège, Fanny Rancière, François Alhenc‐Gelas, Yves Gallois, A Girault, Frédéric Fumeron, Michel Marre, Ronan Roussel, Fabrice Bonnet, Amélie Bonnefond, Stéphane Cauchi, Philippe Froguel, Joël Cogneau, C. Born, E Cacès, M. Cailleau, Olivier Lantieri, J.G. Moreau, F Rakotozafy, Jean Tichet, Sylviane Vol, Michelle Agee, Babak Alipanahi, Adam Auton, Robert K. Bell, Katarzyna Bryc, Sarah L. Elson, Pierre Fontanillas, Nicholas A. Furlotte, Bethann S. Hromatka, Karen E. Huber, Aaron Kleinman, Nadia K. Litterman, Matthew H. McIntyre, Joanna L. Mountain, Carrie A. M. Northover, Steven J. Pitts, J. Fah Sathirapongsasuti, Olga V. Sazonova, Janie F. Shelton, Suyash Shringarpure, Chao Tian, Joyce Y. Tung, Vladimir Vacic, Catherine H. Wilson

Bibliographic record

VenueThe Lancet Neurology · 2017
Typereview
Languageen
FieldMedicine
TopicRestless Legs Syndrome Research
Canadian institutionsUniversity of OttawaMontreal Neurological Institute and HospitalCanadian Sleep & Circadian NetworkHôpital du Sacré-Cœur de MontréalInstitut Universitaire en Santé Mentale de QuébecUniversité de MontréalInstitut Universitaire de Gériatrie de MontréalMcGill University
FundersUCB PharmaHelmholtz Zentrum MünchenNational Institutes of HealthBritannia PharmaceuticalsH. Lundbeck A/SServierNIHR Cambridge Biomedical Research CentreElse Kröner-Fresenius-StiftungDeutsche ForschungsgemeinschaftUniversity of ThessalyEuropean CommissionResMedCovis PharmaEesti TeadusagentuurNational Institute of Neurological Disorders and StrokeBritish Heart FoundationWellcome TrustTeva Pharmaceutical IndustriesMedical Research CouncilPfizerBiogenVifor PharmaRestless Legs Syndrome FoundationUniversity of OxfordNational Institute for Health and Care ResearchNHS Blood and TransplantEuropean Regional Development FundBundesministerium für Bildung und Forschung
KeywordsGenome-wide association studyMeta-analysisOdds ratioGeneticsGenetic associationRestless legs syndromeBiologyCandidate geneLinkage disequilibriumGeneSingle-nucleotide polymorphismBioinformaticsMedicineAlleleHaplotypeNeuroscienceGenotypeInternal medicineNeurology

Abstract

fetched live from OpenAlex

BACKGROUND: Restless legs syndrome is a prevalent chronic neurological disorder with potentially severe mental and physical health consequences. Clearer understanding of the underlying pathophysiology is needed to improve treatment options. We did a meta-analysis of genome-wide association studies (GWASs) to identify potential molecular targets. METHODS: ) were tested for replication in an independent GWAS of 30 770 cases and 286 913 controls, followed by a joint analysis of the discovery and replication stages. We did gene annotation, pathway, and gene-set-enrichment analyses and studied the genetic correlations between restless legs syndrome and traits of interest. FINDINGS: We identified and replicated 13 new risk loci for restless legs syndrome and confirmed the previously identified six risk loci. MEIS1 was confirmed as the strongest genetic risk factor for restless legs syndrome (odds ratio 1·92, 95% CI 1·85-1·99). Gene prioritisation, enrichment, and genetic correlation analyses showed that identified pathways were related to neurodevelopment and highlighted genes linked to axon guidance (associated with SEMA6D), synapse formation (NTNG1), and neuronal specification (HOXB cluster family and MYT1). INTERPRETATION: Identification of new candidate genes and associated pathways will inform future functional research. Advances in understanding of the molecular mechanisms that underlie restless legs syndrome could lead to new treatment options. We focused on common variants; thus, additional studies are needed to dissect the roles of rare and structural variations. FUNDING: Deutsche Forschungsgemeinschaft, Helmholtz Zentrum München-Deutsches Forschungszentrum für Gesundheit und Umwelt, National Research Institutions, NHS Blood and Transplant, National Institute for Health Research, British Heart Foundation, European Commission, European Research Council, National Institutes of Health, National Institute of Neurological Disorders and Stroke, NIH Research Cambridge Biomedical Research Centre, and UK Medical Research Council.

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)0.015
metaresearch head score (Gemma)0.019
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Meta-analysis · Consensus signal: Meta-analysis
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.015
Threshold uncertainty score0.078

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0150.019
Meta-epidemiology (narrow)0.0030.001
Meta-epidemiology (broad)0.0080.032
Bibliometrics0.0050.008
Science and technology studies0.0010.001
Scholarly communication0.0040.001
Open science0.0020.002
Research integrity0.0020.002
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 head0.565
GPT teacher head0.505
Teacher spread0.060 · 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 designMeta-analysis
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".

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Citations240
Published2017
Admission routes1
Has abstractyes

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