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Record W4414997569 · doi:10.1038/s41380-025-03280-x

Cortical microstructure is associated with disease severity and clinical progression in genetic frontotemporal dementia: a GENFI study

2025· article· en· W4414997569 on OpenAlexafffund
Elena Rodriguez‐Vieitez, Melissa Taheri Rydell, Abbe Ullgren, Víctor Montal, Ignacio Illán‐Gala, Juan Fortea, Vesna Jelić, Arabella Bouzigues, Lucy L. Russell, Phoebe H. Foster, Eve Ferry‐Bolder, Lize C. Jiskoot, Harro Seelaar, Raquel Sánchez‐Valle, Robert Laforce, Daniela Galimberti, Rik Vandenberghe, Alexandre de Mendonça, Pietro Tiraboschi, Isabel Santana, Alexander Gerhard, Johannes Levin, Sandro Sorbi, Markus Otto, Florence Pasquier, Simon Ducharme, Christopher Butler, Elizabeth Finger, Maria Carmela Tartaglia, Mario Masellis, James B. Rowe, Matthis Synofzik, Fermín Moreno, Barbara Borroni, Jonathan D. Rohrer, Eric Westman, Caroline Graff

Bibliographic record

VenueMolecular Psychiatry · 2025
Typearticle
Languageen
FieldMedicine
TopicAlzheimer's disease research and treatments
Canadian institutionsSunnybrook Health Science CentreOccupational Cancer Research CentreWestern UniversityMcGill UniversityDouglas Mental Health University InstituteUniversity of TorontoMontreal Neurological Institute and HospitalUniversité Laval
FundersNIHR Cambridge Biomedical Research CentreInstituto de Salud Carlos IIICanadian Institutes of Health ResearchFaculté de Médecine, Université LavalDemensfondenGeorge Institute for Global HealthCentro de Investigación Biomédica en Red sobre Enfermedades NeurodegenerativasUniversitat Autònoma de BarcelonaStichting DioraphteMedical Research CouncilUniversità degli Studi di BresciaEuropean Regional Development FundUniversitat de BarcelonaUniversidade de LisboaÅhlén-stiftelsenFondazione I.R.C.C.S. Istituto Neurologico Carlo BestaEU Joint Programme – Neurodegenerative Disease ResearchUniversità degli Studi di MilanoUniversità degli Studi di FirenzeVetenskapsrådetCambridge University HospitalsStockholms Läns LandstingKU LeuvenUniversidade de CoimbraImperial College LondonUniversity of TorontoBundesministerium für Bildung und ForschungMinisterio de Economía y CompetitividadNederlandse Organisatie voor Wetenschappelijk OnderzoekNational Institute for Health and Care ResearchAlzheimer SocietyInstitut National de la Santé et de la Recherche MédicaleMinistero della SaluteAlzheimer NederlandEberhard Karls Universität TübingenZonMwUniversity of CambridgeSorbonne UniversitéMcGill UniversityAlzheimer's AssociationGlobal Brain Health InstituteWeston Brain InstituteAlzheimerfondenKarolinska InstitutetUniversité de LilleSunnybrook Research InstituteUniversité LavalGun och Bertil Stohnes StiftelseNeuroförbundetStiftelsen för Gamla TjänarinnorBarcelona Supercomputing CenterDeutsches Zentrum für Neurodegenerative Erkrankungen
KeywordsFrontotemporal dementiaC9orf72Clinical Dementia RatingDementiaTauopathyDiseaseAlzheimer's diseaseNeuroimaging

Abstract

fetched live from OpenAlex

The study of genetic frontotemporal dementia (FTD) allows investigating its earliest presymptomatic stages. Using cross-sectional T1-weighted and diffusion-weighted MRI, we test the hypothesis that cortical microstructural alterations, quantified as cortical mean diffusivity (cMD), are detectable earlier and are more strongly associated with clinical progression than cortical thickness (CTh). The sample comprised n = 710 individuals (47.8 ± 13.5 years, 56.6% female, 14.1 ± 3.3 years of education), including 118 symptomatic carriers and 305 presymptomatic carriers with mutations in C9orf72, GRN or MAPT genes, and 287 non-carriers, collected from 24 GENFI sites. A subset of n = 453 individuals (289 carriers, 164 non-carriers) were investigated across Clinical Dementia Rating (CDR) = 0, 0.5 and ≥1 stages. Two subsets had longitudinal clinical outcome measures, including n = 403 individuals (239 carriers, 164 non-carriers) with Cambridge Behavioural Inventory-Revised scores during 2.8 ± 1.6 years, and n = 261 individuals (164 carriers, 97 non-carriers) with CDR Sum-of-Boxes scores during 2.0 ± 0.8 years. Regional cMD and CTh were entered into linear mixed-effects models incorporating age, sex and education as covariates; site, and individual nested within site were random intercepts. The results demonstrated that cMD is more sensitive than CTh to track early cortical injury, with elevated cMD first observed at CDR = 0 in C9orf72 carriers, followed by MAPT carriers (from CDR = 0.5 stage), and by GRN carriers (beginning at CDR ≥ 1). At all stages, cortical microstructural injury had stronger effect size and was more widespread than cortical thinning. In all mutation carrier types, cMD was more strongly associated than CTh with subsequent clinical progression. Cortical microstructure is a promising biomarker to identify at-risk individuals before atrophy and clinical progression, with utility in therapeutic trials.

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)0.002
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 score0.015
Threshold uncertainty score0.031

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0010.001
Science and technology studies0.0010.001
Scholarly communication0.0010.000
Open science0.0010.001
Research integrity0.0010.001
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 head0.352
Teacher spread0.342 · 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
Published2025
Admission routes2
Has abstractyes

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