The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study
| Autor(a) principal: | |
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| Data de Publicação: | 2023 |
| Outros Autores: | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
| Tipo de documento: | Artigo |
| Idioma: | eng |
| Título da fonte: | Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) |
| Texto Completo: | http://hdl.handle.net/10451/56763 |
Resumo: | © 2023 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by- nc-nd/4.0/) |
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The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI studyCognitionFrontotemporal dementiaGeneticMarkerNeuropsychologyPresymptomatic© 2023 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by- nc-nd/4.0/)Objective: Sensitive cognitive markers are still needed for frontotemporal dementia (FTD). The Benson Complex Figure Test (BCFT) is an interesting candidate test, as it assesses visuospatial, visual memory, and executive abilities, allowing the detection of multiple mechanisms of cognitive impairment. To investigate differences in BCFT Copy, Recall and Recognition in presymptomatic and symptomatic FTD mutation carriers, and to explore its cognitive and neuroimaging correlates. Method: We included cross-sectional data from 332 presymptomatic and 136 symptomatic mutation carriers (GRN, MAPT or C9orf72 mutations), and 290 controls in the GENFI consortium. We examined gene-specific differences between mutation carriers (stratified by CDR® NACC-FTLD score) and controls using Quade's / Pearson Χ2 tests. We investigated associations with neuropsychological test scores and grey matter volume using partial correlations and multiple regression models respectively. Results: No significant differences were found between groups at CDR® NACC-FTLD 0-0.5. Symptomatic GRN and C9orf72 mutation carriers had lower Copy scores at CDR® NACC-FTLD ≥2. All three groups had lower Recall scores at CDR® NACC-FTLD ≥2, with MAPT mutation carriers starting at CDR® NACC-FTLD ≥1. All three groups had lower Recognition scores at CDR® NACC FTLD ≥2. Performance correlated with tests for visuoconstruction, memory, and executive function. Copy scores correlated with frontal-subcortical grey matter atrophy, while Recall scores correlated with temporal lobe atrophy. Conclusions: In the symptomatic stage, the BCFT identifies differential mechanisms of cognitive impairment depending on the genetic mutation, corroborated by gene-specific cognitive and neuroimaging correlates. Our findings suggest that impaired performance on the BCFT occurs relatively late in the genetic FTD disease process. Therefore its potential as cognitive biomarker for upcoming clinical trials in presymptomatic to early-stage FTD is most likely limited.The Dementia Research Centre is supported by Alzheimer's Research UK, Alzheimer's Society, Brain Research UK, and The Wolfson Foundation. This work was supported by the NIHR UCL/H Biomedical Research Centre, the Leonard Wolfson Experimental Neurology Centre (LWENC) Clinical Research Facility, and the UK Dementia Research Institute, which receives its funding from UK DRI Ltd., funded by the UK Medical Research Council, Alzheimer's Society and Alzheimer's Research UK. JDR is supported by the Miriam Marks Brain Research UK Senior Fellowship and has received funding from an MRC Clinician Scientist Fellowship (MR/M008525/1) and the NIHR Rare Disease Translational Research Collaboration (BRC149/NS/MH). This work was also supported by the MRC UK GENFI grant (MR/M023664/1), the Bluefield Project, the JPND GENFI-PROX grant (2019–02248), the Dioraphte Foundation [grant numbers 09–02-00], the Association for Frontotemporal Dementias Research Grant 2009, The Netherlands Organization for Scientific Research (NWO) (grant HCMI 056–13-018), ZonMw Memorabel (Deltaplan Dementie; project numbers 733050103 and 733050813), JPND PreFrontAls Consortium (project number 733051042) and Instituto de Salud Carlos III, Spain, and FEDER funds (grant number 20/00448). JBR is supported by the Wellcome Trust (103838), Medical Research Council (SUAG092 G116768) and the NIHR Cambridge Biomedical Research Centre (BRC-1215 − 20014: the views expressed are those of the authors and not necessarily those of the NIHR or the Department of Health and Social Care). This work was funded by the Deutsche Forschungsgemeinschaft (DFG, German Research Foundation) under Germany's Excellence Strategy within the framework of the Munich Cluster for Systems Neurology (EXC 2145 SyNergy – ID 390857198). JMP is supported by a fellowship award from Alzheimer Nederland (WE.15–2019.02). This work was conducted using the MRC Dementias Platform UK (MR/L023784/1 and MR/009076/1).ElsevierRepositório da Universidade de LisboaJiskoot, Lize C.Russell, Lucy L.Peakman, GeorgiaConvery, Rhian S.Greaves, Caroline V.Bocchetta, MartinaPoos, Jackie M.Seelaar, HarroGiannini, Lucia A. A.van Swieten, John C.van Minkelen, RickPijnenburg, Yolande A. L.Rowe, James B.Borroni, BarbaraGalimberti, DanielaMasellis, MarioTartaglia, CarmelaFinger, ElizabethButler, Chris R.Graff, CarolineLaforce, RobertSanchez-Valle, RaquelDe Mendonça, AlexandreMoreno, FerminSynofzik, MatthisVandenberghe, RikDucharme, Simonle Ber, IsabelleLevin, JohannesOtto, MarkusPasquier, FlorenceSantana, IsabelCash, David M.Thomas, DavidRohrer, Jonathan D.2023-03-22T14:57:51Z20232023-01-01T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttp://hdl.handle.net/10451/56763engJ Neurol Sci. 2023 Mar 15;446:1205900022-510X10.1016/j.jns.2023.1205901878-5883info:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2024-11-20T18:20:19Zoai:repositorio.ul.pt:10451/56763Portal AgregadorONGhttps://www.rcaap.pt/oai/openairemluisa.alvim@gmail.comopendoar:71602024-11-20T18:20:19Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse |
| dc.title.none.fl_str_mv |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| title |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| spellingShingle |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study Jiskoot, Lize C. Cognition Frontotemporal dementia Genetic Marker Neuropsychology Presymptomatic |
| title_short |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| title_full |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| title_fullStr |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| title_full_unstemmed |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| title_sort |
The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: a GENFI study |
| author |
Jiskoot, Lize C. |
| author_facet |
Jiskoot, Lize C. Russell, Lucy L. Peakman, Georgia Convery, Rhian S. Greaves, Caroline V. Bocchetta, Martina Poos, Jackie M. Seelaar, Harro Giannini, Lucia A. A. van Swieten, John C. van Minkelen, Rick Pijnenburg, Yolande A. L. Rowe, James B. Borroni, Barbara Galimberti, Daniela Masellis, Mario Tartaglia, Carmela Finger, Elizabeth Butler, Chris R. Graff, Caroline Laforce, Robert Sanchez-Valle, Raquel De Mendonça, Alexandre Moreno, Fermin Synofzik, Matthis Vandenberghe, Rik Ducharme, Simon le Ber, Isabelle Levin, Johannes Otto, Markus Pasquier, Florence Santana, Isabel Cash, David M. Thomas, David Rohrer, Jonathan D. |
| author_role |
author |
| author2 |
Russell, Lucy L. Peakman, Georgia Convery, Rhian S. Greaves, Caroline V. Bocchetta, Martina Poos, Jackie M. Seelaar, Harro Giannini, Lucia A. A. van Swieten, John C. van Minkelen, Rick Pijnenburg, Yolande A. L. Rowe, James B. Borroni, Barbara Galimberti, Daniela Masellis, Mario Tartaglia, Carmela Finger, Elizabeth Butler, Chris R. Graff, Caroline Laforce, Robert Sanchez-Valle, Raquel De Mendonça, Alexandre Moreno, Fermin Synofzik, Matthis Vandenberghe, Rik Ducharme, Simon le Ber, Isabelle Levin, Johannes Otto, Markus Pasquier, Florence Santana, Isabel Cash, David M. Thomas, David Rohrer, Jonathan D. |
| author2_role |
author author author author author author author author author author author author author author author author author author author author author author author author author author author author author author author author author author |
| dc.contributor.none.fl_str_mv |
Repositório da Universidade de Lisboa |
| dc.contributor.author.fl_str_mv |
Jiskoot, Lize C. Russell, Lucy L. Peakman, Georgia Convery, Rhian S. Greaves, Caroline V. Bocchetta, Martina Poos, Jackie M. Seelaar, Harro Giannini, Lucia A. A. van Swieten, John C. van Minkelen, Rick Pijnenburg, Yolande A. L. Rowe, James B. Borroni, Barbara Galimberti, Daniela Masellis, Mario Tartaglia, Carmela Finger, Elizabeth Butler, Chris R. Graff, Caroline Laforce, Robert Sanchez-Valle, Raquel De Mendonça, Alexandre Moreno, Fermin Synofzik, Matthis Vandenberghe, Rik Ducharme, Simon le Ber, Isabelle Levin, Johannes Otto, Markus Pasquier, Florence Santana, Isabel Cash, David M. Thomas, David Rohrer, Jonathan D. |
| dc.subject.por.fl_str_mv |
Cognition Frontotemporal dementia Genetic Marker Neuropsychology Presymptomatic |
| topic |
Cognition Frontotemporal dementia Genetic Marker Neuropsychology Presymptomatic |
| description |
© 2023 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by- nc-nd/4.0/) |
| publishDate |
2023 |
| dc.date.none.fl_str_mv |
2023-03-22T14:57:51Z 2023 2023-01-01T00:00:00Z |
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info:eu-repo/semantics/publishedVersion |
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info:eu-repo/semantics/article |
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article |
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publishedVersion |
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http://hdl.handle.net/10451/56763 |
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http://hdl.handle.net/10451/56763 |
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eng |
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eng |
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J Neurol Sci. 2023 Mar 15;446:120590 0022-510X 10.1016/j.jns.2023.120590 1878-5883 |
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info:eu-repo/semantics/openAccess |
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openAccess |
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application/pdf |
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Elsevier |
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Elsevier |
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Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação |
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