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Record W3015522591 · doi:10.1136/jnnp-2019-322493

Neuronal pentraxin 2: a synapse-derived CSF biomarker in genetic frontotemporal dementia

2020· article· en· W3015522591 on OpenAlexaff
Emma L. van der Ende, Mei-Fang Xiao, Desheng Xu, Jackie M. Poos, Jessica Panman, Lize C. Jiskoot, Lieke Meeter, Elise G.P. Dopper, Janne M. Papma, Carolin Heller, Rhian S. Convery, Katrina Moore, Martina Bocchetta, Mollie Neason, Georgia Peakman, David M. Cash, Charlotte E. Teunissen, Caroline Graff, Matthis Synofzik, Fermín Moreno, Elizabeth Finger, Raquel Sánchez‐Valle, Rik Vandenberghe, Robert Laforce, Mario Masellis, Maria Carmela Tartaglia, James B. Rowe, Christopher Butler, Simon Ducharme, Alexander Gerhard, Adrian Danek, Johannes Levin, Yolande A.L. Pijnenburg, Barbara Borroni, Fabrizio Tagliavini, Alexandre de Mendonça, Isabel Santana, Daniela Galimberti, Harro Seelaar, Jonathan D. Rohrer, Paul F. Worley, John C. van Swieten

Bibliographic record

VenueJournal of Neurology Neurosurgery & Psychiatry · 2020
Typearticle
Languageen
FieldNeuroscience
TopicCerebrospinal fluid and hydrocephalus
Canadian institutionsMcGill UniversityOccupational Cancer Research CentreHealth Sciences CentreMontreal Neurological Institute and HospitalUniversity of TorontoUniversité LavalMcGill University Health CentreSunnybrook Health Science CentreWestern University
FundersNational Institute of Neurological Disorders and StrokeNational Institute on AgingMedical Research CouncilAlzheimer NederlandVetenskapsrådetStockholms Läns LandstingHjärnfondenDeutsche ForschungsgemeinschaftStiftelsen för Gamla TjänarinnorZonMwWellcome TrustNederlandse Organisatie voor Wetenschappelijk OnderzoekAlzheimer's SocietyHorizon 2020 Framework ProgrammeStichting DioraphteWellcome
KeywordsFrontotemporal dementiaC9orf72BiomarkerDementiaCerebrospinal fluidSynapseOncologyMedicineInternal medicineDiseaseNeurosciencePathologyBiologyGenetics

Abstract

fetched live from OpenAlex

INTRODUCTION: Synapse dysfunction is emerging as an early pathological event in frontotemporal dementia (FTD), however biomarkers are lacking. We aimed to investigate the value of cerebrospinal fluid (CSF) neuronal pentraxins (NPTXs), a family of proteins involved in homeostatic synapse plasticity, as novel biomarkers in genetic FTD. METHODS: , and 70 healthy non-carriers participating in the Genetic Frontotemporal dementia Initiative (GENFI), all of whom had at least one CSF sample. We measured CSF concentrations of NPTX2 using an in-house ELISA, and NPTX1 and NPTX receptor (NPTXR) by Western blot. We correlated NPTX2 with corresponding clinical and neuroimaging datasets as well as with CSF neurofilament light chain (NfL) using linear regression analyses. RESULTS: Symptomatic mutation carriers had lower NPTX2 concentrations (median 643 pg/mL, IQR (301-872)) than presymptomatic carriers (1003 pg/mL (624-1358), p<0.001) and non-carriers (990 pg/mL (597-1373), p<0.001) (corrected for age). Similar results were found for NPTX1 and NPTXR. Among mutation carriers, NPTX2 concentration correlated with several clinical disease severity measures, NfL and grey matter volume of the frontal, temporal and parietal lobes, insula and whole brain. NPTX2 predicted subsequent decline in phonemic verbal fluency and Clinical Dementia Rating scale plus FTD modules. In longitudinal CSF samples, available in 13 subjects, NPTX2 decreased around symptom onset and in the symptomatic stage. DISCUSSION: We conclude that NPTX2 is a promising synapse-derived disease progression biomarker in genetic FTD.

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 score0.003
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.000
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.030
GPT teacher head0.253
Teacher spread0.223 · 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

Citations103
Published2020
Admission routes1
Has abstractyes

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