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Record W4213246082 · doi:10.1126/scitranslmed.abj0264

Genome-wide study of DNA methylation shows alterations in metabolic, inflammatory, and cholesterol pathways in ALS

2022· review· en· W4213246082 on OpenAlexafffund
Paul J. Hop, Ramona A.J. Zwamborn, Eilís Hannon, Gemma Shireby, Marta F. Nabais, Emma Walker, Wouter van Rheenen, Joke J.F.A. van Vugt, Annelot M. Dekker, Henk‐Jan Westeneng, Gijs H.P. Tazelaar, Kristel R. van Eijk, Matthieu Moisse, Denis Baird, Ahmad Al Khleifat, Alfredo Iacoangeli, Nicola Ticozzi, Antonia Ratti, Johnathan Cooper‐Knock, Karen Morrison, Pamela J. Shaw, A. Nazlı Başak, Adriano Chiò, Andrea Calvo, Cristina Moglia, Antonio Canosa, Maura Brunetti, Maurizio Grassano, Marc Gotkine, Yossef Lerner, Michal Zabari, Patrick Vourc’h, Philippe Corcia, P. Couratier, Jesús S. Mora Pardina, Teresa Salas, Patrick A. Dion, Jay P. Ross, Robert D. Henderson, Susan Mathers, Pamela A. McCombe, Merrilee Needham, Garth Nicholson, Dominic B. Rowe, Roger Pamphlett, Karen A. Mather, Perminder S. Sachdev, Sarah Furlong, Fleur C. Garton, Anjali K. Henders, Tian Lin, Shyuan T. Ngo, Frederik J. Steyn, Leanne Wallace, Kelly L. Williams, Miguel Mitne Neto, Ruben J. Cauchi, Ian P. Blair, Matthew C. Kiernan, Vivian Drory, Mónica Povedano, Mamede de Carvalho, Susana Pinto, Markus Weber, Guy A. Rouleau, Vincenzo Silani, John E. Landers, Christopher E. Shaw, Peter M. Andersen, Allan F. McRae, Michael A. van Es, R. Jeroen Pasterkamp, Naomi R. Wray, Russell L. McLaughlin, Orla Hardiman, Kevin P. Kenna, Ellen Tsai, Heiko Runz, Ammar Al‐Chalabi, Leonard H. van den Berg, Philip Van Damme, Jonathan Mill, Jan H. Veldink, Bastiaan T. Heijmans, Peter A.C. t Hoen, Joyce van Meurs, Rick Jansen, Lude Franke, Dorret I. Boomsma, René Pool, Jenny van Dongen, Joukje J. Hottenga, Marleen M. J. van Greevenbroek, Coen D.A. Stehouwer, Carla Kallen, Casper G. Schalkwijk, Cisca Wijmenga, Ettje F. Tigchelaar, P. Eline Slagboom, Marian Beekman, Joris Deelen, Jan H. Veldink, Cornelia M. van Duijn, Bert A. Hofman, Aaron Isaacs, André G. Uitterlinden, Joyce van Meurs, P. Mila Jhamai, Michaël Verbiest, H. Eka D. Suchiman, Marijn Verkerk, Ruud van der Breggen, Jeroen van Rooij, Nico Lakenberg, Hailiang Mei, Maarten van Iterson, Michiel van Galen, Jan Bot, Dasha V. Zhernakova, Peter van ‘t Hof, Patrick Deelen, Irene Nooren, Matthijs Moed, Martijn Vermaat, René Luijk, Marc Jan Bonder, Freerk van Dijk, Wibowo Arindrarto, Szymon M. Kielbasa, Morris A. Swertz, Erik W. van Zwet, Gilbert Bensimon, George Davey Smith

Bibliographic record

VenueScience Translational Medicine · 2022
Typereview
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsMcGill UniversityMontreal Neurological Institute and Hospital
FundersNational Institute of Neurological Disorders and StrokeNational Health and Medical Research CouncilEconomic and Social Research CouncilMedical Research CouncilMitsubishi Tanabe Pharma CorporationVlaamse regeringKoç ÜniversitesiPrinses Beatrix SpierfondsKnut och Alice Wallenbergs StiftelseKU LeuvenMinistero dell’Istruzione, dell’Università e della RicercaNederlandse Organisatie voor Wetenschappelijk OnderzoekCanadian Institutes of Health ResearchFonds Wetenschappelijk OnderzoekKing's College LondonCSL BehringNational Institute for Health and Care ResearchAlexion PharmaceuticalsApellis PharmaceuticalsArgenxEU Joint Programme – Neurodegenerative Disease ResearchUlla-Carin Lindquists stiftelse för ALS-forskningHealth~HollandRegeneron PharmaceuticalsVetenskapsrådetE-RareCytokineticsMotor Neurone Disease Research Institute of AustraliaEli Lilly and CompanyStichting ALS NederlandAgence Nationale de la RechercheMinistero della SaluteAlzheimer's SocietyWellcome TrustMotor Neurone Disease AssociationNIHR Maudsley Biomedical Research CentreVästerbotten Läns LandstingSanofiSouth London and Maudsley NHS Foundation TrustBiogenFightMNDUmeå UniversitetEuropean Commission
KeywordsDNA methylationBiologyEpigenomeMissing heritability problemGeneticsMethylationGenome-wide association studyCholesterolDiseaseGeneBioinformaticsMedicineInternal medicineEndocrinologyGenotypeGene expressionSingle-nucleotide polymorphism

Abstract

fetched live from OpenAlex

Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease with an estimated heritability between 40 and 50%. DNA methylation patterns can serve as proxies of (past) exposures and disease progression, as well as providing a potential mechanism that mediates genetic or environmental risk. Here, we present a blood-based epigenome-wide association study meta-analysis in 9706 samples passing stringent quality control (6763 patients, 2943 controls). We identified a total of 45 differentially methylated positions (DMPs) annotated to 42 genes, which are enriched for pathways and traits related to metabolism, cholesterol biosynthesis, and immunity. We then tested 39 DNA methylation-based proxies of putative ALS risk factors and found that high-density lipoprotein cholesterol, body mass index, white blood cell proportions, and alcohol intake were independently associated with ALS. Integration of these results with our latest genome-wide association study showed that cholesterol biosynthesis was potentially causally related to ALS. Last, DNA methylation at several DMPs and blood cell proportion estimates derived from DNA methylation data were associated with survival rate in patients, suggesting that they might represent indicators of underlying disease processes potentially amenable to therapeutic interventions.

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)0.004
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.923
Threshold uncertainty score0.992

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0040.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0020.000
Bibliometrics0.0020.003
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.000

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 head0.130
GPT teacher head0.388
Teacher spread0.259 · 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.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
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

Citations93
Published2022
Admission routes2
Has abstractyes

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