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Record W3215312374 · doi:10.1186/s13024-021-00499-4

A panel of CSF proteins separates genetic frontotemporal dementia from presymptomatic mutation carriers: a GENFI study

2021· article· en· W3215312374 on OpenAlex

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.
fundA Canadian funder is recorded on the work.

Bibliographic record

VenueMolecular Neurodegeneration · 2021
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsMcGill UniversityDouglas Mental Health University InstituteUniversité LavalHealth Sciences CentreOccupational Cancer Research CentreMontreal Neurological Institute and HospitalUniversity of TorontoSunnybrook Health Science CentreWestern University
FundersRobarts Research InstituteMedical Research CouncilDemensfondenStichting DioraphteUniversität UlmUniversità degli Studi di BresciaUniversidade de LisboaÅhlén-stiftelsenCentre National de la Recherche ScientifiqueUniversità degli Studi di FirenzeVetenskapsrådetStockholms Läns LandstingKU LeuvenFamiljen Erling-Perssons StiftelseUniversity Health NetworkHjärnfondenUniversidade de CoimbraUniversity of TorontoNederlandse Organisatie voor Wetenschappelijk OnderzoekEberhard Karls Universität TübingenKing's College LondonZonMwKarolinska InstitutetSunnybrook Research InstituteEU Joint Programme – Neurodegenerative Disease ResearchInstitut National de la Santé et de la Recherche MédicaleKungliga Tekniska HögskolanErasmus Medisch CentrumBrain Research UKBundesministerium für Bildung und ForschungLudwig-Maximilians-Universität MünchenNational Institute for Health and Care ResearchSorbonne UniversitéUniversità degli Studi di MilanoMcGill UniversityFondazione I.R.C.C.S. Istituto Neurologico Carlo BestaStiftelsen för Gamla Tjänarinnor
KeywordsFrontotemporal dementiaC9orf72MutationDementiaPathologicalMedicineBiologyBioinformaticsPathologyGeneticsDiseaseGene

Abstract

fetched live from OpenAlex

BACKGROUND: A detailed understanding of the pathological processes involved in genetic frontotemporal dementia is critical in order to provide the patients with an optimal future treatment. Protein levels in CSF have the potential to reflect different pathophysiological processes in the brain. We aimed to identify and evaluate panels of CSF proteins with potential to separate symptomatic individuals from individuals without clinical symptoms (unaffected), as well as presymptomatic individuals from mutation non-carriers. METHODS: A multiplexed antibody-based suspension bead array was used to analyse levels of 111 proteins in CSF samples from 221 individuals from families with genetic frontotemporal dementia. The data was explored using LASSO and Random forest. RESULTS: When comparing affected individuals with unaffected individuals, 14 proteins were identified as potentially important for the separation. Among these, four were identified as most important, namely neurofilament medium polypeptide (NEFM), neuronal pentraxin 2 (NPTX2), neurosecretory protein VGF (VGF) and aquaporin 4 (AQP4). The combined profile of these four proteins successfully separated the two groups, with higher levels of NEFM and AQP4 and lower levels of NPTX2 in affected compared to unaffected individuals. VGF contributed to the models, but the levels were not significantly lower in affected individuals. Next, when comparing presymptomatic GRN and C9orf72 mutation carriers in proximity to symptom onset with mutation non-carriers, six proteins were identified with a potential to contribute to a separation, including progranulin (GRN). CONCLUSION: In conclusion, we have identified several proteins with the combined potential to separate affected individuals from unaffected individuals, as well as proteins with potential to contribute to the separation between presymptomatic individuals and mutation non-carriers. Further studies are needed to continue the investigation of these proteins and their potential association to the pathophysiological mechanisms 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.

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: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.489
Threshold uncertainty score0.767

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 head0.287
Teacher spread0.256 · 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