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Record W4306391018 · doi:10.1002/acn3.51672

<scp>CSF</scp> glial markers are elevated in a subset of patients with genetic frontotemporal dementia

2022· article· en· W4306391018 on OpenAlexafffund
Ione Woollacott, Imogen J. Swift, Aitana Sogorb‐Esteve, Carolin Heller, Kathryn Knowles, Arabella Bouzigues, Lucy L. Russell, Georgia Peakman, Caroline Greaves, Rhian S. Convery, Amanda Heslegrave, James B. Rowe, Barbara Borroni, Daniela Galimberti, Pietro Tiraboschi, Mario Masellis, Maria Carmela Tartaglia, Elizabeth Finger, John C. van Swieten, Harro Seelaar, Lize C. Jiskoot, Sandro Sorbi, Christopher Butler, Caroline Graff, Alexander Gerhard, Robert Laforce, Raquel Sánchez‐Valle, Alexandre de Mendonça, Fermín Moreno, Matthis Synofzik, Rik Vandenberghe, Simon Ducharme, Isabelle Le Ber, Johannes Levin, Markus Otto, Florence Pasquier, Isabel Santana, Henrik Zetterberg, Jonathan D. Rohrer

Bibliographic record

VenueAnnals of Clinical and Translational Neurology · 2022
Typearticle
Languageen
FieldNeuroscience
TopicNeuroinflammation and Neurodegeneration Mechanisms
Canadian institutionsMcGill UniversityDouglas Mental Health University InstituteUniversité LavalHealth Sciences CentreOccupational Cancer Research CentreMontreal Neurological Institute and HospitalUniversity of TorontoWestern UniversitySunnybrook Health Science Centre
FundersH2020 Marie Skłodowska-Curie ActionsNIHR Cambridge Biomedical Research CentreEuropean Research CouncilCanadian Institutes of Health ResearchTau ConsortiumOlav Thon StiftelsenAlzheimer NederlandStichting DioraphteUniversiteit HasseltUniversität UlmMedical Research CouncilMedical Research Council CanadaDeutsches Zentrum für Neurodegenerative ErkrankungenVetenskapsrådetStockholms Läns LandstingKU LeuvenCentro de Investigación Biomédica en Red sobre Enfermedades NeurodegenerativasCentre National de la Recherche ScientifiqueWolfson FoundationFamiljen Erling-Perssons StiftelseEuropean CommissionUniversity of CambridgeBundesministerium für Bildung und ForschungNational Institute for Health and Care ResearchUniversidad Internacional de La RiojaNational Brain AppealNederlandse Organisatie voor Wetenschappelijk OnderzoekAgence Nationale de la RechercheWellcome TrustMinistero della SaluteAlzheimer's SocietyDeutsche ForschungsgemeinschaftWeston Brain InstituteZonMwAlzheimer's Drug Discovery FoundationBrain Research UKUniversidad de La RiojaInstitut National de la Santé et de la Recherche MédicaleEU Joint Programme – Neurodegenerative Disease ResearchUniversiteit AntwerpenStiftelsen för Gamla TjänarinnorAlzheimer's AssociationUK Dementia Research InstituteOntario Brain Institute
KeywordsFrontotemporal dementiaTREM2MedicineCerebrospinal fluidC9orf72Internal medicineOncologyMutationNeuroinflammationDementiaConfidence intervalGastroenterologyPathologyDiseaseMicrogliaInflammationGeneticsBiologyGene

Abstract

fetched live from OpenAlex

Abstract Background Neuroinflammation has been shown to be an important pathophysiological disease mechanism in frontotemporal dementia (FTD). This includes activation of microglia, a process that can be measured in life through assaying different glia‐derived biomarkers in cerebrospinal fluid. However, only a few studies so far have taken place in FTD, and even fewer focusing on the genetic forms of FTD. Methods We investigated the cerebrospinal fluid concentrations of TREM2, YKL‐40 and chitotriosidase using immunoassays in 183 participants from the Genetic FTD Initiative (GENFI) study: 49 C9orf72 (36 presymptomatic, 13 symptomatic), 49 GRN (37 presymptomatic, 12 symptomatic) and 23 MAPT (16 presymptomatic, 7 symptomatic) mutation carriers and 62 mutation‐negative controls. Concentrations were compared between groups using a linear regression model adjusting for age and sex, with 95% bias‐corrected bootstrapped confidence intervals. Concentrations in each group were correlated with the Mini‐Mental State Examination (MMSE) score using non‐parametric partial correlations adjusting for age. Age‐adjusted z‐scores were also created for the concentration of markers in each participant, investigating how many had a value above the 95th percentile of controls. Results Only chitotriosidase in symptomatic GRN mutation carriers had a concentration significantly higher than controls. No group had higher TREM2 or YKL‐40 concentrations than controls after adjusting for age and sex. There was a significant negative correlation of chitotriosidase concentration with MMSE in presymptomatic GRN mutation carriers. In the symptomatic groups, for TREM2 31% of C9orf72, 25% of GRN, and 14% of MAPT mutation carriers had a concentration above the 95th percentile of controls. For YKL‐40 this was 8% C9orf72, 8% GRN and 0% MAPT mutation carriers, whilst for chitotriosidase it was 23% C9orf72, 50% GRN, and 29% MAPT mutation carriers. Conclusions Although chitotriosidase concentrations in GRN mutation carriers were the only significantly raised glia‐derived biomarker as a group, a subset of mutation carriers in all three groups, particularly for chitotriosidase and TREM2, had elevated concentrations. Further work is required to understand the variability in concentrations and the extent of neuroinflammation across the genetic forms of FTD. However, the current findings suggest limited utility of these measures in forthcoming 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)0.000
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 score0.004
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0010.000
Open science0.0000.000
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 head0.074
GPT teacher head0.316
Teacher spread0.241 · 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".

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Citations13
Published2022
Admission routes2
Has abstractyes

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