MétaCan
Menu
Back to cohort
Record W3147351247 · doi:10.1212/wnl.0000000000011848

Plasma Neurofilament Light for Prediction of Disease Progression in Familial Frontotemporal Lobar Degeneration

2021· article· en· W3147351247 on OpenAlexafffund
Julio C. Rojas, Ping Wang, Adam M. Staffaroni, Carolin Heller, Yann Cobigo, Amy Wolf, Sheng‐Yang M. Goh, Peter A. Ljubenkov, Hilary W. Heuer, Jamie Fong, Joanne Taylor, Eliseo Veras, Linan Song, Andreas Jeromin, David Hanlon, Lili Yu, Arvind Khinikar, Rajeev Sivasankaran, Agnieszka Kieloch, Marie‐Anne Valentin, Anna M. Karydas, Laura L. Mitic, Rodney Pearlman, John Kornak, Joel H. Kramer, Bruce L. Miller, Kejal Kantarci, David S. Knopman, Neill R. Graff‐Radford, Leonard Petrucelli, Rosa Rademakers, David J. Irwin, Murray Grossman, Eliana Marisa Ramos, Giovanni Coppola, Mario F. Mendez, Yvette Bordelon, Bradford C. Dickerson, Nupur Ghoshal, Edward D. Huey, Ian R. Mackenzie, Brian S. Appleby, Kimiko Domoto‐Reilly, Ging‐Yuek Robin Hsiung, Arthur W. Toga, Sandra Weıntraub, Daniel Kaufer, Diana Kerwin, Irene Litvan, Chiadikaobi Onyike, Alexander Pantelyat, Erik D. Roberson, Maria Carmela Tartaglia, Tatiana Foroud, Weiping Chen, Julie Czerkowicz, Danielle Graham, John C. van Swieten, Barbara Borroni, Raquel Sánchez‐Valle, Fermín Moreno, Robert Laforce, Caroline Graff, Matthis Synofzik, Daniela Galimberti, James B. Rowe, Mario Masellis, Elizabeth Finger, Rik Vandenberghe, Alexandre de Mendonça, Fabrizio Tagliavini, Isabel Santana, Simon Ducharme, Christopher Butler, Alexander Gerhard, Johannes Levin, Adrian Danek, Markus Otto, Sandro Sorbi, David M. Cash, Rhian S. Convery, Martina Bocchetta, Martha Foiani, Caroline Greaves, Georgia Peakman, Lucy L. Russell, Imogen J. Swift, Emily Todd, Jonathan D. Rohrer, Bradley F. Boeve, Howard J. Rosen, Adam L. Boxer

Bibliographic record

VenueNeurology · 2021
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsUniversity of TorontoUniversity of British ColumbiaWestern University
FundersNational Institute of Neurological Disorders and StrokeNIHR Cambridge Biomedical Research CentreJanssen PharmaceuticalsAlzheimer Society of B.C.Medical Research CouncilCanadian Institutes of Health ResearchAvid RadiopharmaceuticalsNational Institutes of HealthUK Dementia Research InstituteEisaiVetenskapsrådetNational Institute of Mental HealthStockholms Läns LandstingIonis PharmaceuticalsWolfson FoundationNational Institute on AgingNational Institute for Health and Care ResearchLarry L. Hillblom FoundationGenentechAstraZenecaNational Brain AppealWellcome TrustAlzheimer SocietyAlzheimer's SocietyDeutsche ForschungsgemeinschaftEli Lilly and CompanyBrain Research UKTauRx PharmaceuticalsKarolinska InstitutetEU Joint Programme – Neurodegenerative Disease ResearchLittle Family FoundationBiogen
KeywordsAsymptomaticFrontotemporal lobar degenerationMedicineInternal medicineAsymptomatic carrierFrontotemporal dementiaOncologyDementiaC9orf72DiseaseGastroenterologyPathology

Abstract

fetched live from OpenAlex

OBJECTIVE: We tested the hypothesis that plasma neurofilament light chain (NfL) identifies asymptomatic carriers of familial frontotemporal lobar degeneration (FTLD)-causing mutations at risk of disease progression. METHODS: mutation carriers and noncarriers from the same families were classified by disease severity (asymptomatic, prodromal, and full phenotype) using the CDR Dementia Staging Instrument plus behavior and language domains from the National Alzheimer's Disease Coordinating Center FTLD module (CDR+NACC-FTLD). Linear mixed-effect models related NfL to clinical variables. RESULTS: = 0.035). Plasma NfL discriminated symptomatic from asymptomatic mutation carriers or those with prodromal disease (original cutoff: 13.6 pg/mL, 87.5% sensitivity, 82.7% specificity; validation cutoff: 19.8 pg/mL, 87.4% sensitivity, 84.3% specificity). Higher baseline NfL correlated with worse longitudinal CDR+NACC-FTLD sum of boxes scores, neuropsychological function, and atrophy, regardless of genotype or disease severity, including asymptomatic mutation carriers. CONCLUSIONS: Plasma NfL identifies asymptomatic carriers of FTLD-causing mutations at short-term risk of disease progression and is a potential tool to select participants for prevention clinical trials. TRIAL REGISTRATION INFORMATION: ClinicalTrials.gov Identifier: NCT02372773 and NCT02365922. CLASSIFICATION OF EVIDENCE: This study provides Class I evidence that in carriers of FTLD-causing mutations, elevation of plasma NfL predicts short-term risk of clinical progression.

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.549
Threshold uncertainty score0.316

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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 head0.031
GPT teacher head0.304
Teacher spread0.273 · 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 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

Citations104
Published2021
Admission routes2
Has abstractyes

Explore more

Same venueNeurologySame topicAmyotrophic Lateral Sclerosis ResearchFrench-language works237,207