The Benson Complex Figure Test detects deficits in visuoconstruction and visual memory in symptomatic familial frontotemporal dementia: A GENFI study
Lize C Jiskoot
(1)
,
Lucy L Russell
(2)
,
Georgia Peakman
(2)
,
Rhian S Convery
(2)
,
Caroline V Greaves
(2)
,
Martina Bocchetta
(2)
,
Jackie M Poos
(1)
,
Harro Seelaar
(1)
,
Lucia A.A. Giannini
(1)
,
John C van Swieten
(1)
,
Rick van Minkelen
(1)
,
Yolande A.L. Pijnenburg
,
James B Rowe
(3)
,
Barbara Borroni
(4)
,
Daniela Galimberti
(5)
,
Mario Masellis
(6)
,
Carmela Tartaglia
(7)
,
Elizabeth Finger
(8)
,
Chris R Butler
(9)
,
Caroline Graff
(10)
,
Robert Laforce
(11)
,
Raquel Sanchez-Valle
(12)
,
Alexandre de Mendonça
(13)
,
Fermin Moreno
,
Matthis Synofzik
(14)
,
Rik Vandenberghe
(15)
,
Simon Ducharme
(16)
,
Isabelle Le Ber
(17)
,
Johannes Levin
(18)
,
Markus Otto
(19)
,
Florence Pasquier
(20)
,
Isabel Santana
(21)
,
David M Cash
(2)
,
David Thomas
(2)
,
Jonathan D Rohrer
(2)
,
Arabella Bouzigues
,
Martin N. Rossor
,
Nick C. Fox
,
Jason D. Warren
,
Imogen J. Swift
,
Rachelle Shafei
,
Carolin Heller
,
Emily Todd
,
Ione Woollacott
,
Henrik Zetterberg
,
Annabel Nelson
,
Rita Guerreiro
,
Jose Bras
,
David L. Thomas
,
Simon Mead Phd
,
Alina Díez Msc
,
Ekaterina Rogaeva
,
Morris Freedman
,
Ron Keren
,
David Tang-Wai
,
Hakan Thonberg
,
Linn Öijerstedt
,
Christin Andersson
,
Vesna Jelic
,
Andrea Arighi
,
Chiara Fenoglio
,
Elio Scarpini
,
Giorgio Fumagalli
,
Thomas Cope
,
Carolyn Timberlake
,
Timothy Rittman
,
Christen Shoesmith
,
Robart Bartha
,
Rosa Rademakers
,
Carlo Wilke
,
Hans-Otto Karnarth
,
Benjamin Bender
,
Rose Bruffaerts
,
Philip Vandamme
,
Mathieu Vandenbulcke
,
Catarina B. Ferreira
,
Gabriel Miltenberger
,
Carolina Maruta
,
Ana Verdelho
,
Sónia Afonso
,
Ricardo Taipa
,
Paola Caroppo
,
Giuseppe Di Fede
,
Giorgio Giaccone
,
Sara Prioni
,
Veronica Redaelli
,
Giacomina Rossi
,
Pietro Tiraboschi
,
Diana Duro
,
Maria Rosario Almeida
,
Miguel Castelo-Branco
,
Maria João Leitão
,
Miguel Tabuas-Pereira
,
Beatriz Santiago
,
Serge Gauthier
,
Pedro Rosa-Neto
,
Michele Veldsman
,
Paul Thompson
,
Tobias Langheinrich
,
Catharina Prix
,
Tobias Hoegen
,
Elisabeth Wlasich
,
Sandra Loosli
,
Sonja Schonecker
,
Sarah Anderl-Straub
,
Jolina Lombardi
,
Nuria Bargalló
,
Alberto Benussi
,
Valentina Cantoni
,
Maxime Bertoux
,
Anne Bertrand
(17)
,
Alexis Brice
(17)
,
Agnès Camuzat
(17)
,
Olivier Colliot
(17)
,
Sabrina Sayah
(17)
,
Aurélie Funkiewiez
(17)
,
Daisy Rinaldi
(17)
,
Gemma Lombardi
(22)
,
Benedetta Nacmias
(22)
,
Dario Saracino
(17)
,
Valentina Bessi
,
Camilla Ferrari
,
Marta Cañada
,
Vincent Deramecourt
,
Gregory Kuchcinski
,
Thibaud Lebouvier
,
Cristina Polito
,
Adeline Rollin
1
Erasmus MC -
Erasmus University Medical Center [Rotterdam]
2 DRC - Dementia Research Centre [London]
3 CAM - University of Cambridge [UK]
4 UniBs - Università degli Studi di Brescia = University of Brescia
5 Centro Dino Ferrari [Milano]
6 SRI - Sunnybrook Research Institute [Toronto]
7 University of Toronto
8 UWO - University of Western Ontario
9 University of Oxford
10 Karolinska University Hospital [Stockholm]
11 ULaval - Université Laval [Québec]
12 IDIBAPS - Institut d'Investigacions Biomèdiques August Pi i Sunyer
13 ULISBOA - Universidade de Lisboa = University of Lisbon = Université de Lisbonne
14 Universitätsklinikum Tübingen - University Hospital of Tübingen
15 KU Leuven - Catholic University of Leuven = Katholieke Universiteit Leuven
16 MUHC - McGill University Health Center [Montreal]
17 ICM - Institut du Cerveau = Paris Brain Institute
18 LMU - Ludwig Maximilian University [Munich] = Ludwig Maximilians Universität München
19 Universitätsklinikum Ulm - University Hospital of Ulm
20 U1172 Inserm - Equipe Alzheimer and Tauopathies - LilNCog
21 UC - University of Coimbra [Portugal]
22 CHU Pitié-Salpêtrière [AP-HP]
2 DRC - Dementia Research Centre [London]
3 CAM - University of Cambridge [UK]
4 UniBs - Università degli Studi di Brescia = University of Brescia
5 Centro Dino Ferrari [Milano]
6 SRI - Sunnybrook Research Institute [Toronto]
7 University of Toronto
8 UWO - University of Western Ontario
9 University of Oxford
10 Karolinska University Hospital [Stockholm]
11 ULaval - Université Laval [Québec]
12 IDIBAPS - Institut d'Investigacions Biomèdiques August Pi i Sunyer
13 ULISBOA - Universidade de Lisboa = University of Lisbon = Université de Lisbonne
14 Universitätsklinikum Tübingen - University Hospital of Tübingen
15 KU Leuven - Catholic University of Leuven = Katholieke Universiteit Leuven
16 MUHC - McGill University Health Center [Montreal]
17 ICM - Institut du Cerveau = Paris Brain Institute
18 LMU - Ludwig Maximilian University [Munich] = Ludwig Maximilians Universität München
19 Universitätsklinikum Ulm - University Hospital of Ulm
20 U1172 Inserm - Equipe Alzheimer and Tauopathies - LilNCog
21 UC - University of Coimbra [Portugal]
22 CHU Pitié-Salpêtrière [AP-HP]
Martina Bocchetta
- Function : Author
- PersonId : 1363148
- ORCID : 0000-0003-1814-5024
John C van Swieten
- Function : Author
- PersonId : 1370402
Yolande A.L. Pijnenburg
- Function : Author
Fermin Moreno
- Function : Author
Matthis Synofzik
- Function : Author
- PersonId : 887987
Rik Vandenberghe
- Function : Author
- PersonId : 921848
Johannes Levin
- Function : Author
- PersonId : 1370407
Markus Otto
- Function : Author
- PersonId : 1370408
Jonathan D Rohrer
- Function : Author
- PersonId : 1110171
- ORCID : 0000-0002-6155-8417
Arabella Bouzigues
- Function : Author
Martin N. Rossor
- Function : Author
Nick C. Fox
- Function : Author
Jason D. Warren
- Function : Author
Imogen J. Swift
- Function : Author
Rachelle Shafei
- Function : Author
Carolin Heller
- Function : Author
Emily Todd
- Function : Author
Ione Woollacott
- Function : Author
Henrik Zetterberg
- Function : Author
Annabel Nelson
- Function : Author
Rita Guerreiro
- Function : Author
Jose Bras
- Function : Author
David L. Thomas
- Function : Author
Simon Mead Phd
- Function : Author
Alina Díez Msc
- Function : Author
Ekaterina Rogaeva
- Function : Author
Morris Freedman
- Function : Author
Ron Keren
- Function : Author
David Tang-Wai
- Function : Author
Hakan Thonberg
- Function : Author
Linn Öijerstedt
- Function : Author
Christin Andersson
- Function : Author
Vesna Jelic
- Function : Author
Andrea Arighi
- Function : Author
Chiara Fenoglio
- Function : Author
Elio Scarpini
- Function : Author
Giorgio Fumagalli
- Function : Author
Thomas Cope
- Function : Author
Carolyn Timberlake
- Function : Author
Timothy Rittman
- Function : Author
Christen Shoesmith
- Function : Author
Robart Bartha
- Function : Author
Rosa Rademakers
- Function : Author
Carlo Wilke
- Function : Author
Hans-Otto Karnarth
- Function : Author
Benjamin Bender
- Function : Author
Rose Bruffaerts
- Function : Author
Philip Vandamme
- Function : Author
Mathieu Vandenbulcke
- Function : Author
Catarina B. Ferreira
- Function : Author
Gabriel Miltenberger
- Function : Author
Carolina Maruta
- Function : Author
Ana Verdelho
- Function : Author
Sónia Afonso
- Function : Author
Ricardo Taipa
- Function : Author
Paola Caroppo
- Function : Author
Giuseppe Di Fede
- Function : Author
Giorgio Giaccone
- Function : Author
Sara Prioni
- Function : Author
Veronica Redaelli
- Function : Author
Giacomina Rossi
- Function : Author
Pietro Tiraboschi
- Function : Author
Diana Duro
- Function : Author
Maria Rosario Almeida
- Function : Author
Miguel Castelo-Branco
- Function : Author
Maria João Leitão
- Function : Author
Miguel Tabuas-Pereira
- Function : Author
Beatriz Santiago
- Function : Author
Serge Gauthier
- Function : Author
Pedro Rosa-Neto
- Function : Author
Michele Veldsman
- Function : Author
Paul Thompson
- Function : Author
Tobias Langheinrich
- Function : Author
Catharina Prix
- Function : Author
Tobias Hoegen
- Function : Author
Elisabeth Wlasich
- Function : Author
Sandra Loosli
- Function : Author
Sonja Schonecker
- Function : Author
Sarah Anderl-Straub
- Function : Author
Jolina Lombardi
- Function : Author
Nuria Bargalló
- Function : Author
Alberto Benussi
- Function : Author
Valentina Cantoni
- Function : Author
Maxime Bertoux
- Function : Author
Alexis Brice
- Function : Author
- PersonId : 1104774
- ORCID : 0000-0002-0941-3990
- IdRef : 050512935
Valentina Bessi
- Function : Author
Camilla Ferrari
- Function : Author
Marta Cañada
- Function : Author
Vincent Deramecourt
- Function : Author
Gregory Kuchcinski
- Function : Author
Thibaud Lebouvier
- Function : Author
- PersonId : 754049
- IdHAL : thibaud-lebouvier
- ORCID : 0000-0002-6630-394X
- IdRef : 177112190
Cristina Polito
- Function : Author
Adeline Rollin
- Function : Author
Abstract
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 Х2 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.
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