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Record W4294647492 · doi:10.1186/s12974-022-02573-0

Elevated CSF and plasma complement proteins in genetic frontotemporal dementia: results from the GENFI study

2022· article· en· W4294647492 on OpenAlexafffund
Emma L. van der Ende, Carolin Heller, Aitana Sogorb‐Esteve, Imogen J. Swift, David McFall, Georgia Peakman, Arabella Bouzigues, Jackie M. Poos, Lize C. Jiskoot, Jessica Panman, Janne M. Papma, Lieke Meeter, Elise G.P. Dopper, Martina Bocchetta, Emily Todd, David M. Cash, 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, Markus Otto, Barbara Borroni, Fabrizio Tagliavini, Alexandre de Mendonça, Isabel Santana, Daniela Galimberti, Sandro Sorbi, Henrik Zetterberg, Eric J. Huang, John C. van Swieten, Jonathan D. Rohrer, Harro Seelaar, Sónia Afonso, Maria Rosário Almeida, Sarah Anderl‐Straub, Christin Andersson, Anna Antonell, Silvana Archetti, Andrea Arighi, Mircea Balasa, Myriam Barandiarán, Núria Bargalló, Benjamin Bender, Alberto Benussi, Luisa Benussi, Valentina Bessi, Giuliano Binetti, Sandra E. Black, Sergi Borrego‐Écija, José Brás, Rose Bruffaerts, Marta Cañada, Valentina Cantoni, Paola Caroppo, Miguel Castelo‐Branco, Rhian S. Convery, Thomas Cope, Giuseppe Di Fede, Alina Díez, Diana Duro, Chiara Fenoglio, Camilla Ferrari, Catarina B. Ferreira, Nick C. Fox, Morris Freedman, Giorgio Fumagalli, Alazne Gabilondo, Roberto Gasparotti, Serge Gauthier, Stefano Gazzina, Giorgio Giaccone, Ana Gorostidi, Caroline Greaves, Rita Guerreiro, Tobias Hoegen, Begoña Indakoetxea, Vesna Jelić, Hans‐Otto Karnath, Ron Keren, Tobias Langheinrich, Maria João Leitão, Albert Lladó, Gemma Lombardi, Sandra Loosli, Carolina Maruta, Simon Mead, Gabriel Miltenberger, Rick van Minkelen, Sara Mitchell, Katrina Moore, Benedetta Nacmias, Jennifer Nicholas, Linn Öijerstedt, Jaume Olives, Sébastien Ourselin, Alessandro Padovani, Michela Pievani, Cristina Polito, Enrico Premi, Sara Prioni, Catharina Prix, Rosa Rademakers, Veronica Redaelli, Timothy Rittman, Ekaterina Rogaeva, Pedro Rosa‐Neto, Giacomina Rossi, Martin Rosser, Beatriz Santiago, Elio Scarpini, Sonja Schönecker, Elisa Semler, Rachelle Shafei, Christen Shoesmith, Miguel Tábuas‐Pereira, Mikel Tainta, Ricardo Taipa, David F. Tang‐Wai, David L. Thomas, Paul Thompson, Håkan Thonberg, Carolyn Timberlake, Pietro Tiraboschi, Philip Van Damme, Mathieu Vandenbulcke, Michele Veldsman, Ana Verdelho, Jorge Villanúa, Jason D. Warren, Carlo Wilke, Ione Woollacott, Elisabeth Wlasich, Miren Zulaica

Bibliographic record

VenueJournal of Neuroinflammation · 2022
Typearticle
Languageen
FieldNeuroscience
TopicNeuroinflammation and Neurodegeneration Mechanisms
Canadian institutionsMcGill University Health CentreMontreal Neurological Institute and HospitalUniversity of TorontoSunnybrook HospitalOccupational Cancer Research CentreUniversité LavalWestern University
FundersInstitute on the Environment, University of MinnesotaMedical Research CouncilDemensfondenUK Dementia Research InstituteStichting DioraphteUniversità degli Studi di BresciaUniversidade de LisboaDeutsches Zentrum für Neurodegenerative ErkrankungenUniversità degli Studi di FirenzeVetenskapsrådetKnut och Alice Wallenbergs StiftelseHorizon 2020 Framework ProgrammeStockholms Läns LandstingKU LeuvenUniversity Health NetworkHjärnfondenUniversidade de CoimbraUniversity of TorontoUniversität UlmToronto Rehabilitation InstituteErasmus Medisch CentrumUniversity of OxfordFondazione IRCCS Ca' Granda Ospedale Maggiore PoliclinicoEberhard Karls Universität TübingenKing's College LondonLudwig-Maximilians-Universität MünchenNational Institute for Health and Care ResearchMcGill UniversityDeutsche ForschungsgemeinschaftBrain Research UKEU Joint Programme – Neurodegenerative Disease ResearchAlzheimer NederlandSunnybrook Research InstituteRobarts Research InstituteAlzheimer's SocietyWellcome TrustGun och Bertil Stohnes StiftelseLondon School of Hygiene and Tropical MedicineUniversità degli Studi di MilanoKarolinska InstitutetZonMwFondazione I.R.C.C.S. Istituto Neurologico Carlo BestaStiftelsen för Gamla TjänarinnorAlzheimer's Association
KeywordsFrontotemporal dementiaC9orf72Frontotemporal lobar degenerationDementiaComplement systemPathologyGrey matterMedicineImmunologyPsychologyDiseaseWhite matterAntibodyMagnetic resonance imaging

Abstract

fetched live from OpenAlex

BACKGROUND: Neuroinflammation is emerging as an important pathological process in frontotemporal dementia (FTD), but biomarkers are lacking. We aimed to determine the value of complement proteins, which are key components of innate immunity, as biomarkers in cerebrospinal fluid (CSF) and plasma of presymptomatic and symptomatic genetic FTD mutation carriers. METHODS: We measured the complement proteins C1q and C3b in CSF by ELISAs in 224 presymptomatic and symptomatic GRN, C9orf72 or MAPT mutation carriers and non-carriers participating in the Genetic Frontotemporal Dementia Initiative (GENFI), a multicentre cohort study. Next, we used multiplex immunoassays to measure a panel of 14 complement proteins in plasma of 431 GENFI participants. We correlated complement protein levels with corresponding clinical and neuroimaging data, neurofilament light chain (NfL) and glial fibrillary acidic protein (GFAP). RESULTS: CSF C1q and C3b, as well as plasma C2 and C3, were elevated in symptomatic mutation carriers compared to presymptomatic carriers and non-carriers. In genetic subgroup analyses, these differences remained statistically significant for C9orf72 mutation carriers. In presymptomatic carriers, several complement proteins correlated negatively with grey matter volume of FTD-related regions and positively with NfL and GFAP. In symptomatic carriers, correlations were additionally observed with disease duration and with Mini Mental State Examination and Clinical Dementia Rating scale® plus NACC Frontotemporal lobar degeneration sum of boxes scores. CONCLUSIONS: Elevated levels of CSF C1q and C3b, as well as plasma C2 and C3, demonstrate the presence of complement activation in the symptomatic stage of genetic FTD. Intriguingly, correlations with several disease measures in presymptomatic carriers suggest that complement protein levels might increase before symptom onset. Although the overlap between groups precludes their use as diagnostic markers, further research is needed to determine their potential to monitor dysregulation of the complement system in 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 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)0.001
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: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.415
Threshold uncertainty score0.590

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.046
GPT teacher head0.261
Teacher spread0.215 · 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 designBench or experimental
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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Citations37
Published2022
Admission routes2
Has abstractyes

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