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Title: | The CBI-R detects early behavioural impairment in genetic frontotemporal dementia |
Author: | Nelson, A Russell, LL Peakman, G Convery, RS Bouzigues, A Greaves, CV Bocchetta, M Cash, DM Swieten, JC Jiskoot, L Moreno, F Sanchez del Valle Díaz, Raquel Laforce, R Graff, C Masellis, M Tartaglia, MC Rowe, JB Borroni, B Finger, E Synofzik, M Galimberti, D Vandenberghe, R Mendona, A Butler, CR Gerhard, A Ducharme, S Le Ber, I Santana, I Pasquier, F Levin, J Otto, M Sorbi, S Rohrer, JD |
Keywords: | Adult Age Alzheimer Disease Amygdala Article Atrophy Behavior behavioural impairment C9orf72 Protein Clinical Assessment Corpus Striatum demographics Disease Severity Education Frontotemporal Dementia Ftld FUNCTIONAL BRAIN CONNECTIVITY Gene Mutation Genetics guanine nucleotide exchange C9orf72 HEXANUCLEOTIDE REPEAT Hippocampus Human Humans Locomotion Mini Mental State Examination Mutation mutation carrier Mutations Neuroimaging Nuclear Magnetic Resonance Imaging Pick Disease of the Brain Pick presenile dementia Progranulins Progression Questionnaire Sex Tau Tau Protein Tau Proteins TEMPORAL VARIANT Thalamus |
Abstract: | Introduction: Behavioural dysfunction is a key feature of genetic frontotemporal dementia (FTD) but validated clinical scales measuring behaviour are lacking at present. Methods: We assessed behaviour using the revised version of the Cambridge Behavioural Inventory (CBI-R) in 733 participants from the Genetic FTD Initiative study: 466 mutation carriers (195 C9orf72, 76 MAPT, 195 GRN) and 267 non-mutation carriers (controls). All mutation carriers were stratified according to their global CDR plus NACC FTLD score into three groups: asymptomatic (CDR = 0), prodromal (CDR = 0.5) and symptomatic (CDR = 1+). Mixed-effects models adjusted for age, education, sex and family clustering were used to compare between the groups. Neuroanatomical correlates of the individual domains were assessed within each genetic group. Results: CBI-R total scores were significantly higher in all CDR 1+ mutation carrier groups compared with controls [C9orf72 mean 70.5 (standard deviation 27.8), GRN 56.2 (33.5), MAPT 62.1 (36.9)] as well as their respective CDR 0.5 groups [C9orf72 13.5 (14.4), GRN 13.3 (13.5), MAPT 9.4 (10.4)] and CDR 0 groups [C9orf72 6.0 (7.9), GRN 3.6 (6.0), MAPT 8.5 (13.3)]. The C9orf72 and GRN 0.5 groups scored significantly higher than the controls. The greatest impairment was seen in the Motivation domain for the C9orf72 and GRN symptomatic groups, whilst in the symptomatic MAPTgroup, the highest-scoring domains were Stereotypic and Motor Behaviours and Memory and Orientation. Neural correlates of each CBI-R domain largely overlapped across the different mutation carrier groups. Conclusions: The CBI-R detects early behavioural change in genetic FTD, suggesting that it could be a useful measure within future clinical trials. |
Note: | Reproducció del document publicat a: https://doi.org/10.1002/acn3.51544 |
It is part of: | Annals Of Clinical And Translational Neurology, 9, 5, 644-658 |
URI: | http://hdl.handle.net/2445/209293 |
Related resource: | https://doi.org/10.1002/acn3.51544 |
ISSN: | Nelson, A;Russell, LL;Peakman, G;Convery, RS;Bouzigues, A;Greaves, CV;Bocchetta, M;Cash, DM;Swieten, JC;Jiskoot, L;Moreno, F;Sanchez del Valle Díaz, Raquel;Laforce, R;Graff, C;Masellis, M;Tartaglia, MC;Rowe, JB;Borroni, B;Finger, E;Synofzik, M;Galimberti, D;Vandenberghe, R;Mendona, A;Butler, CR;Gerhard, A;Ducharme, S;Le Ber, I;Santana, I;Pasquier, F;Levin, J;Otto, M;Sorbi, S;Rohrer, JD. The CBI-R detects early behavioural impairment in genetic frontotemporal dementia. Annals Of Clinical And Translational Neurology, 9, 5, 644-658 |
Appears in Collections: | Articles publicats en revistes (IDIBAPS: Institut d'investigacions Biomèdiques August Pi i Sunyer) |
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