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Record W4309149093 · doi:10.1007/s00415-022-11442-y

Motor symptoms in genetic frontotemporal dementia: developing a new module for clinical rating scales

2022· article· en· W4309149093 on OpenAlexafffund
Kiran Samra, Amy MacDougall, Georgia Peakman, Arabella Bouzigues, Martina Bocchetta, David M. Cash, Caroline Greaves, Rhian S. Convery, John C. van Swieten, Lize C. Jiskoot, Harro Seelaar, Fermín Moreno, Raquel Sánchez‐Valle, Robert Laforce, Caroline Graff, Mario Masellis, Maria Carmela Tartaglia, James B. Rowe, Barbara Borroni, Elizabeth Finger, Matthis Synofzik, Daniela Galimberti, Rik Vandenberghe, Alexandre de Mendonça, Christopher Butler, Alexander Gerhard, Simon Ducharme, Isabelle Le Ber, Pietro Tiraboschi, Isabel Santana, Florence Pasquier, Johannes Levin, Markus Otto, Sandro Sorbi, Jonathan D. Rohrer, Lucy L. Russell, Annabel Nelson, David L. Thomas, Emily Todd, Hanya Benotmane, Jennifer Nicholas, Rachelle Shafei, Carolyn Timberlake, Thomas Cope, Timothy Rittman, Alberto Benussi, Enrico Premi, Roberto Gasparotti, Silvana Archetti, Stefano Gazzina, Valentina Cantoni, Andrea Arighi, Chiara Fenoglio, Giorgio Fumagalli, Vittoria Borracci, Giacomina Rossi, Giorgio Giaccone, Giuseppe Di Fede, Paola Caroppo, Sara Prioni, Veronica Redaelli, David F. Tang‐Wai, Ekaterina Rogaeva, Miguel Castelo‐Branco, Morris Freedman, Ron Keren, Sandra E. Black, Sara Mitchell, Christen Shoesmith, Rosa Rademakers, Jackie M. Poos, Janne M. Papma, Lucia Giannini, Rick van Minkelen, Yolande A.L. Pijnenburg, Benedetta Nacmias, Camilla Ferrari, Cristina Polito, Gemma Lombardi, Valentina Bessi, Michele Veldsman, Christin Andersson, Håkan Thonberg, Linn Öijerstedt, Vesna Jelić, Paul Thompson, Tobias Langheinrich, Albert Lladó, Anna Antonell, Jaume Olives, Mircea Balasa, Núria Bargalló, Sergi Borrego‐Écija, Ana Verdelho, Carolina Maruta, Catarina B. Ferreira, Gabriel Miltenberger, Frederico Simões do Couto, Alazne Gabilondo, Ana Gorostidi, Jorge Villanúa, Marta Cañada, Mikel Tainta, Miren Zulaica, Myriam Barandiarán, Patricia Alves, Benjamin Bender, Carlo Wilke, Lisa Graf, Annick Vogels, Mathieu Vandenbulcke, Philip Van Damme, Rose Bruffaerts, Koen Poesen, Pedro Rosa‐Neto, Serge Gauthier, Agnès Camuzat, Alexis Brice, Anne Bertrand, Aurélie Funkiewiez, Daisy Rinaldi, Dario Saracino, Olivier Colliot, Sabrina Sayah, Catharina Prix, Elisabeth Wlasich, Olivia Wagemann, Sandra Loosli, Sonja Schönecker, Tobias Hoegen, Jolina Lombardi, Sarah Anderl‐Straub, Adeline Rollin, Grégory Kuchcinski, Maxime Bertoux, Thibaud Lebouvier, Vincent Deramecourt, Beatriz Santiago, Diana Duro, Maria João Leitão, Maria Rosário Almeida, Miguel Tábuas‐Pereira, Sónia Afonso

Bibliographic record

VenueJournal of Neurology · 2022
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsMcGill UniversityWestern UniversityMontreal Neurological Institute and HospitalUniversity of TorontoOccupational Cancer Research CentreHealth Sciences CentreMcGill University Health CentreSunnybrook Health Science CentreUniversité Laval
FundersNIHR Cambridge Biomedical Research CentreInstituto de Salud Carlos IIIMedical Research CouncilUK Dementia Research InstituteStichting DioraphteFondazione IRCCS Ca' Granda Ospedale Maggiore PoliclinicoFondazione I.R.C.C.S. Istituto Neurologico Carlo BestaUniversiteit AntwerpenUniversity of TorontoUniversidade de CoimbraUniversità degli Studi di BresciaRobarts Research InstituteAlzheimer's SocietyMinistero della SaluteToronto Rehabilitation InstituteCentro de Investigación Biomédica en Red sobre Enfermedades NeurodegenerativasWolfson FoundationWellcome TrustUniversity College LondonNational Institute for Health and Care ResearchBrain Research UKAlzheimer's Research TrustNational Brain AppealNederlandse Organisatie voor Wetenschappelijk OnderzoekErasmus Medisch CentrumDeutsche ForschungsgemeinschaftUniversity Health NetworkUniversità degli Studi di MilanoEU Joint Programme – Neurodegenerative Disease ResearchSunnybrook Research InstituteFundació la Marató de TV3Weston Brain InstituteCanadian Institutes of Health ResearchLondon School of Hygiene and Tropical Medicine
KeywordsFrontotemporal dementiaC9orf72Clinical Dementia RatingParkinsonismFrontotemporal lobar degenerationRating scaleAmyotrophic lateral sclerosisDementiaPsychologyMedicineDysarthriaPhysical medicine and rehabilitationExecutive dysfunctionInternal medicinePsychiatryNeuropsychologyDiseaseCognition

Abstract

fetched live from OpenAlex

Abstract Objective To investigate the optimal method of adding motor features to a clinical rating scale for frontotemporal dementia (FTD). Methods Eight hundred and thirty-two participants from the international multicentre Genetic FTD Initiative (GENFI) study were recruited: 522 mutation carriers (with C9orf72 , GRN and MAPT mutations) and 310 mutation-negative controls. A standardised clinical questionnaire was used to assess eight motor symptoms (dysarthria, dysphagia, tremor, slowness, weakness, gait disorder, falls and functional difficulties using hands). Frequency and severity of each motor symptom was assessed, and a principal component analysis (PCA) was performed to identify how the different motor symptoms loaded together. Finally, addition of a motor component to the CDR ® plus NACC FTLD was investigated (CDR ® plus NACC FTLD-M). Results 24.3% of mutation carriers had motor symptoms (31.7% C9orf72 , 18.8% GRN , 19.3% MAPT ) compared to 6.8% of controls. Slowness and gait disorder were the commonest in all genetic groups while tremor and falls were the least frequent. Symptom severity scores were similar to equivalent physical motor examination scores. PCA revealed that all motor symptoms loaded together so a single additional motor component was added to the CDR ® plus NACC FTLD to form the CDR ® plus NACC FTLD-M. Individual global scores were more severe with the CDR ® plus NACC FTLD-M, and no patients with a clinically diagnosed motor disorder (ALS/FTD-ALS or parkinsonism) were classified anymore as asymptomatic (unlike the CDR ® plus NACC FTLD alone). Conclusions Motor features are present in mutation carriers at all disease stages across all three genetic groups. Inclusion of motor symptoms in a rating scale that can be used in future clinical trials will not only ensure a more accurate severity measure is recorded but that a wider spectrum of FTD phenotypes can be included in the same trial.

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)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.015
Threshold uncertainty score0.331

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
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 integrity0.0000.001
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 head0.089
GPT teacher head0.387
Teacher spread0.298 · 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".

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Citations18
Published2022
Admission routes2
Has abstractyes

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