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Record W4399735224 · doi:10.1016/j.ajhg.2024.05.017

Genome-wide analyses reveal a potential role for the MAPT, MOBP, and APOE loci in sporadic frontotemporal dementia

2024· article· en· W4399735224 on OpenAlexafffund
Claudia Manzoni, Demis A. Kia, Raffaele Ferrari, Ganna Leonenko, Beatrice Costa, Valentina Saba, Edwin Jabbari, Manuela Tan, Diego Albani, Victoria Álvarez, Ignacio Álvarez, Ole A. Andreassen, Antonella Angiolillo, Andrea Arighi, Matt Baker, Luisa Benussi, Valentina Bessi, Giuliano Binetti, D. Blackburn, Merçé Boada, Bradley F. Boeve, Sergi Borrego‐Écija, Barbara Borroni, Geir Bråthen, William S. Brooks, Amalia C. Bruni, Paola Caroppo, Sara Bandrés‐Ciga, Jordi Clarimón, Rosanna Colao, Carlos Cruchaga, Adrian Danek, Sterre CM. de Boer, Itziar de Rojas, Alfonso Di Costanzo, Dennis W. Dickson, Janine Diehl‐Schmid, Carol Dobson‐Stone, Oriol Dols‐Icardo, Aldo Donizetti, Elise G.P. Dopper, Elisabetta Durante, Camilla Ferrari, Gianluigi Forloni, Francesca Frangipane, Laura Fratiglioni, Milica G. Kramberger, Daniela Galimberti, Maurizio Gallucci, Pablo García‐González, Roberta Ghidoni, Giorgio Giaccone, Caroline Graff, Neill R. Graff‐Radford, Jordan Grafman, Glenda M. Halliday, Dena Hernandez, Lena E. Hjermind, John R. Hodges, Guy Holloway, Edward D. Huey, Ignacio Illán‐Gala, Keith A. Josephs, David S. Knopman, Mark Kristiansen, John B. Kwok, Isabelle Leber, Hampton L. Leonard, Ilenia Libri, Alberto Lleó, Ian R. Mackenzie, Gaganjit K. Madhan, Raffaele Maletta, Marta Marquié, Aleš Maver, Manuel Menéndez‐González, Graziella Milan, Bruce L. Miller, Christopher M. Morris, Huw R. Morris, Benedetta Nacmias, Judith Newton, Jørgen E. Nielsen, Christer Nilsson, Valeria Novelli, Alessandro Padovani, Suvankar Pal, Florence Pasquier, Pau Pástor, Robert Perneczky, Borut Peterlin, Ronald C. Petersen, Olivier Piguet, Yolande A.L. Pijnenburg, Annibale Alessandro Puca, Rosa Rademakers, Innocenzo Rainero, Lianne M. Reus, Anna MT. Richardson, Ekaterina Rogaeva, Boris Rogelj, Sara Rollinson, Howard Rosen, Giacomina Rossi, James B. Rowe, Elisa Rubino, Agustı́n Ruiz, Erika Salvi, Raquel Sánchez‐Valle, Sigrid Botne Sando, Alexander Santillo, Jennifer A. Saxon, Johannes C. M. Schlachetzki, Sonja W. Scholz, Harro Seelaar, William W. Seeley, María Serpente, Sandro Sorbi, Sabrina Sordon, Peter St George‐Hyslop, J. C. Thompson, Christine Van Broeckhoven, Vivianna M. Van Deerlin, Sven J Van Der Lee, John C. van Swieten, Fabrizio Tagliavini, Julie van der Zee, Arianna Veronesi, Emilia Vitale, Maria Landqvist Waldö, Jennifer S. Yokoyama, Mike A. Nalls, Parastoo Momeni, Andrew Singleton, John Hardy, Valentina Escott‐Price

Bibliographic record

VenueThe American Journal of Human Genetics · 2024
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsOccupational Cancer Research CentreYork UniversityUniversity of TorontoUniversity of British ColumbiaVancouver Coastal Health
FundersCilagNIHR Cambridge Biomedical Research CentreEuropean Regional Development FundInstituto de Salud Carlos IIICanadian Institutes of Health ResearchGrifolsCentro de Investigación Biomédica en Red sobre Enfermedades NeurodegenerativasNorges ForskningsrådDeutsches Zentrum für Neurodegenerative ErkrankungenNovo NordiskKyowa Hakko KirinEisaiRocheDemensförbundetDeutsche ForschungsgemeinschaftMinisterio de Ciencia e InnovaciónEuropean CommissionMedical Research CouncilDemensfondenNeuraxpharmDepartment of Health and Social CareConsortium canadien en neurodégénérescence associée au vieillissementWellcome TrustHope Center for Neurological DisordersUK Dementia Research InstituteEuropean Federation of Pharmaceutical Industries and AssociationsEU Joint Programme – Neurodegenerative Disease ResearchNational Institute on AgingNational Institute for Health and Care ResearchPSP AssociationBiogenNational Health and Medical Research CouncilInternational Parkinson and Movement Disorder SocietyNovo Nordisk FondenNewcastle UniversityCurePSPAgentschap Innoveren en OndernemenMinistero della SaluteStiftung VERUMAlzheimer's SocietyNational Institutes of HealthAssociation for Frontotemporal DegenerationNasjonalforeningen for FolkehelsenMichael J. Fox Foundation for Parkinson's ResearchEli Lilly and CompanyU.S. Department of Health and Human Services
KeywordsFrontotemporal dementiaDementiaDiseaseMedicinePsychologyPathology

Abstract

fetched live from OpenAlex

Frontotemporal dementia (FTD) is the second most common cause of early-onset dementia after Alzheimer disease (AD). Efforts in the field mainly focus on familial forms of disease (fFTDs), while studies of the genetic etiology of sporadic FTD (sFTD) have been less common. In the current work, we analyzed 4,685 sFTD cases and 15,308 controls looking for common genetic determinants for sFTD. We found a cluster of variants at the MAPT (rs199443; p = 2.5 × 10 −12 , OR = 1.27) and APOE (rs6857; p = 1.31 × 10 −12 , OR = 1.27) loci and a candidate locus on chromosome 3 (rs1009966; p = 2.41 × 10 −8 , OR = 1.16) in the intergenic region between RPSA and MOBP , contributing to increased risk for sFTD through effects on expression and/or splicing in brain cortex of functionally relevant in- cis genes at the MAPT and RPSA - MOBP loci. The association with the MAPT (H1c clade) and RPSA - MOBP loci may suggest common genetic pleiotropy across FTD and progressive supranuclear palsy (PSP) ( MAPT and RPSA - MOBP loci) and across FTD, AD, Parkinson disease (PD), and cortico-basal degeneration (CBD) ( MAPT locus). Our data also suggest population specificity of the risk signals, with MAPT and APOE loci associations mainly driven by Central/Nordic and Mediterranean Europeans, respectively. This study lays the foundations for future work aimed at further characterizing population-specific features of potential FTD-discriminant APOE haplotype(s) and the functional involvement and contribution of the MAPT H1c haplotype and RPSA - MOBP loci to pathogenesis of sporadic forms of FTD in brain cortex.

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)0.001
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 score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0010.000
Research integrity0.0010.000
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 head0.041
GPT teacher head0.352
Teacher spread0.311 · 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

Citations14
Published2024
Admission routes2
Has abstractyes

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