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Examining empathy deficits across familial forms of frontotemporal dementia within the GENFI cohort

2022Artigo científico Acesso aberto
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dc.contributor.authorFoster, Phoebe H.
dc.contributor.authorRussell, Lucy L.
dc.contributor.authorPeakman, Georgia
dc.contributor.authorConvery, Rhian S.
dc.contributor.authorBouzigues, Arabella
dc.contributor.authorGreaves, Caroline V.
dc.contributor.authorBocchetta, Martina
dc.contributor.authorCash, David M.
dc.contributor.authorvan Swieten, John C.
dc.contributor.authorJiskoot, Lize C.
dc.contributor.authorMoreno, Fermin
dc.contributor.authorSanchez-Valle, Raquel
dc.contributor.authorLaforce, Robert
dc.contributor.authorGraff, Caroline
dc.contributor.authorMasellis, Mario
dc.contributor.authorTartaglia, Carmela
dc.contributor.authorRowe, James B.
dc.contributor.authorBorroni, Barbara
dc.contributor.authorFinger, Elizabeth
dc.contributor.authorSynofzik, Matthis
dc.contributor.authorGalimberti, Daniela
dc.contributor.authorVandenberghe, Rik
dc.contributor.authorDe Mendonça, Alexandre
dc.contributor.authorButler, Chris R.
dc.contributor.authorGerhard, Alex
dc.contributor.authorDucharme, Simon
dc.contributor.authorLe Ber, Isabelle
dc.contributor.authorTagliavini, Fabrizio
dc.contributor.authorSantana, Isabel
dc.contributor.authorPasquier, Florence
dc.contributor.authorLevin, Johannes
dc.contributor.authorDanek, Adrian
dc.contributor.authorOtto, Markus
dc.contributor.authorSorbi, Sandro
dc.contributor.authorRohrer, Jonathan D.
dc.contributor.authorAfonso, Sónia
dc.contributor.authorAlmeida, Maria Rosario
dc.contributor.authorAnderl-Straub, Sarah
dc.contributor.authorAndersson, Christin
dc.contributor.authorAntonell, Anna
dc.contributor.authorArchetti, Silvana
dc.contributor.authorArighi, Andrea
dc.contributor.authorBalasa, Mircea
dc.contributor.authorBarandiaran, Myriam
dc.contributor.authorBargalló, Nuria
dc.contributor.authorBartha, Robart
dc.contributor.authorBender, Benjamin
dc.contributor.authorBenussi, Alberto
dc.contributor.authorBertoux, Maxime
dc.contributor.authorBertrand, Anne
dc.contributor.authorBessi, Valentina
dc.contributor.authorBlack, Sandra
dc.contributor.authorBorrego-Ecija, Sergi
dc.contributor.authorBras, Jose
dc.contributor.authorBrice, Alexis
dc.contributor.authorBruffaerts, Rose
dc.contributor.authorCamuzat, Agnès
dc.contributor.authorCañada, Marta
dc.contributor.authorCantoni, Valentina
dc.contributor.authorCaroppo, Paola
dc.contributor.authorCash, David
dc.contributor.authorCastelo-Branco, Miguel
dc.contributor.authorColliot, Olivier
dc.contributor.authorCope, Thomas
dc.contributor.authorDeramecourt, Vincent
dc.contributor.authorde Arriba, María
dc.contributor.authorDi Fede, Giuseppe
dc.contributor.authorDíez, Alina
dc.contributor.authorDuro, Diana
dc.contributor.authorFenoglio, Chiara
dc.contributor.authorFerrari, Camilla
dc.contributor.authorFerreira, Catarina B.
dc.contributor.authorFox, Nick
dc.contributor.authorFreedman, Morris
dc.contributor.authorFumagalli, Giorgio
dc.contributor.authorFunkiewiez, Aurélie
dc.contributor.authorGabilondo, Alazne
dc.contributor.authorGasparotti, Roberto
dc.contributor.authorGauthier, Serge
dc.contributor.authorGazzina, Stefano
dc.contributor.authorGiaccone, Giorgio
dc.contributor.authorGorostidi, Ana
dc.contributor.authorGreaves, Caroline
dc.contributor.authorGuerreiro, Rita
dc.contributor.authorHeller, Carolin
dc.contributor.authorHoegen, Tobias
dc.contributor.authorIndakoetxea, Begoña
dc.contributor.authorJelic, Vesna
dc.contributor.authorKarnath, Hans-Otto
dc.contributor.authorKeren, Ron
dc.contributor.authorKuchcinski, Gregory
dc.contributor.authorLangheinrich, Tobias
dc.contributor.authorLebouvier, Thibaud
dc.contributor.authorLeitão, Maria João
dc.contributor.authorLladó, Albert
dc.contributor.authorLombardi, Gemma
dc.contributor.authorLoosli, Sandra
dc.contributor.authorMaruta, Carolina
dc.contributor.authorMead, Simon
dc.contributor.authorMeeter, Lieke
dc.contributor.authorMiltenberger-Miltenyi, Gabriel
dc.contributor.authorvan Minkelen, Rick
dc.contributor.authorMitchell, Sara
dc.contributor.authorMoore, Katrina
dc.contributor.authorNacmias, Benedetta
dc.contributor.authorNelson, Annabel
dc.contributor.authorÖijerstedt, Linn
dc.contributor.authorOlives, Jaume
dc.contributor.authorOurselin, Sebastien
dc.contributor.authorPadovani, Alessandro
dc.contributor.authorPanman, Jessica
dc.contributor.authorPapma, Janne M.
dc.contributor.authorPijnenburg, Yolande
dc.contributor.authorPolito, Cristina
dc.contributor.authorPremi, Enrico
dc.contributor.authorPrioni, Sara
dc.contributor.authorPrix, Catharina
dc.contributor.authorRademakers, Rosa
dc.contributor.authorRedaelli, Veronica
dc.contributor.authorRinaldi, Daisy
dc.contributor.authorRittman, Tim
dc.contributor.authorRogaeva, Ekaterina
dc.contributor.authorRollin, Adeline
dc.contributor.authorRosa-Neto, Pedro
dc.contributor.authorRossi, Giacomina
dc.contributor.authorRossor, Martin
dc.contributor.authorSantiago, Beatriz
dc.contributor.authorSaracino, Dario
dc.contributor.authorSayah, Sabrina
dc.contributor.authorScarpini, Elio
dc.contributor.authorSchönecker, Sonja
dc.contributor.authorSeelaar, Harro
dc.contributor.authorSemler, Elisa
dc.contributor.authorShafei, Rachelle
dc.contributor.authorShoesmith, Christen
dc.contributor.authorSwift, Imogen
dc.contributor.authorTábuas-Pereira, Miguel
dc.contributor.authorTainta, Mikel
dc.contributor.authorTaipa, Ricardo
dc.contributor.authorTang-Wai, David
dc.contributor.authorThomas, David L.
dc.contributor.authorThompson, Paul
dc.contributor.authorThonberg, Hakan
dc.contributor.authorTimberlake, Carolyn
dc.contributor.authorTiraboschi, Pietro
dc.contributor.authorTodd, Emily
dc.contributor.authorVan Damme, Philip
dc.contributor.authorVandenbulcke, Mathieu
dc.contributor.authorVeldsman, Michele
dc.contributor.authorVerdelho, Ana
dc.contributor.authorVillanua, Jorge
dc.contributor.authorWarren, Jason
dc.contributor.authorWilke, Carlo
dc.contributor.authorWoollacott, Ione
dc.contributor.authorWlasich, Elisabeth
dc.contributor.authorZetterberg, Henrik
dc.contributor.authorZulaica, Miren
dc.date.accessioned2022-05-03T13:28:32Z
dc.date.available2022-05-03T13:28:32Z
dc.date.issued2022
dc.description© 2022 The Author(s). Published by Elsevier Ltd. This is an open access article under the CC BY license (http:// creativecommons.org/licenses/by/4.0/).pt_PT
dc.description.abstractBackground: Reduced empathy is a common symptom in frontotemporal dementia (FTD). Although empathy deficits have been extensively researched in sporadic cases, few studies have explored the differences in familial forms of FTD. Methods: Empathy was examined using a modified version of the Interpersonal Reactivity Index (mIRI) in 676 participants from the Genetic FTD Initiative: 216 mutation-negative controls, 192 C9orf72 expansion carriers, 193 GRN mutation carriers and 75 MAPT mutation carriers. Using global scores from the CDR® plus NACC FTLD, mutation carriers were divided into three groups, asymptomatic (0), very mildly symptomatic/prodromal (.5), or fully symptomatic (1 or more). The mIRI Total score, as well as the subscores of Empathic Concern (EC) and Perspective Taking (PT) were assessed. Linear regression models with bootstrapping were used to assess empathy ratings across genetic groups, as well as across phenotypes in the symptomatic carriers. Neural correlates of empathy deficits were examined using a voxel-based morphometry (VBM) analysis. Results: All fully symptomatic groups scored lower on the mIRI Total, EC, and PT when compared to controls and their asymptomatic or prodromal counterparts (all p < .001). Prodromal C9orf72 expansion carriers also scored significantly lower than controls on the mIRI Total score (p = .046). In the phenotype analysis, all groups (behavioural variant FTD, primary progressive aphasia and FTD with amyotrophic lateral sclerosis) scored significantly lower than controls (all p < .007). VBM revealed an overlapping neural correlate of the mIRI Total score across genetic groups in the orbitofrontal lobe but with additional involvement in the temporal lobe, insula and basal ganglia in both the GRN and MAPT groups, and uniquely more posterior regions such as the parietal lobe and thalamus in the GRN group, and medial temporal structures in the MAPT group. Conclusions: Significant empathy deficits present in genetic FTD, particularly in symptomatic individuals and those with a bvFTD phenotype, while prodromal deficits are only seen using the mIRI in C9orf72 expansion carriers.pt_PT
dc.description.sponsorshipThis 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 an MRC Clinician Scientist Fellowship (MR/M008525/1) and has received funding from 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 and the JPND GENFI-PROX grant (2019-02248). Several authors of this publication are members of the European Reference Network for Rare Neurological Diseases - Project ID No 739510. RC/CG are supported by a Frontotemporal Dementia Research Studentships in Memory of David Blechner funded through The National Brain Appeal (RCN 290173). MB is supported by a Fellowship award from the Alzheimer's Society, UK (AS-JF-19a-004-517). MB's work is also supported by the UK Dementia Research Institute which receives its funding from DRI Ltd, funded by the UK Medical Research Council, Alzheimer's Society and Alzheimer's Research UK. JCVS was supported by the Dioraphte Foundation grant 09-02-03-00, the Association for Frontotemporal Dementias Research Grant 2009, The Netherlands Organisation for Scientific Research grant HCMI 056-13-018, ZonMw Memorabel (Deltaplan Dementie, project number 733 051 042), Alzheimer Nederland and the Bluefield project. FM received funding from the Tau Consortium and the Center for Networked Biomedical Research on Neurodegenerative Disease (CIBERNED). RS-V is supported by an Alzheimer’s Research UK Clinical Research Training Fellowship (ARUK-CRF2017B-2), and has received funding from Fundació Marató de TV3, Spain (grant no. 20143810). CG received funding from JPND-Prefrontals VR Dnr 529-2014-7504, VR 2015-02926 and 2018-02754, the Swedish FTD Inititative-Schörling Foundation, Alzheimer Foundation, Brain Foundation and Stockholm County Council ALF. MM has received funding from a Canadian Institute of Health Research operating grant and the Weston Brain Institute and Ontario Brain Institute. JBR has received funding from the Wellcome Trust (103838) and is supported by the Cambridge University Centre for Frontotemporal Dementia, the Medical Research Council (SUAG/051 G101400) and the National Institute for Health Research Cambridge Biomedical Research Centre (BRC-1215-20014). EF has received funding from a CIHR grant #327387. DG received support from the EU Joint Programme – Neurodegenerative Disease Research and the Italian Ministry of Health (PreFrontALS) grant 733051042. RV has received funding from the Mady Browaeys Fund for Research into Frontotemporal Dementia. MO has received funding from BMBF (FTLDc).pt_PT
dc.description.versioninfo:eu-repo/semantics/publishedVersionpt_PT
dc.identifier.citationCortex. 2022 May;150:12-28pt_PT
dc.identifier.doi10.1016/j.cortex.2022.01.012pt_PT
dc.identifier.eissn1973-8102
dc.identifier.issn0010-9452
dc.identifier.urihttp://hdl.handle.net/10451/52646
dc.language.isoengpt_PT
dc.peerreviewedyespt_PT
dc.publisherElsevierpt_PT
dc.relation.publisherversionhttps://www.sciencedirect.com/journal/cortexpt_PT
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/pt_PT
dc.subjectEmpathic concernpt_PT
dc.subjectEmpathypt_PT
dc.subjectFrontotemporal dementiapt_PT
dc.subjectInterpersonal Reactivity Indexpt_PT
dc.subjectPerspective takingpt_PT
dc.titleExamining empathy deficits across familial forms of frontotemporal dementia within the GENFI cohortpt_PT
dc.typejournal article
dspace.entity.typePublication
oaire.citation.endPage28pt_PT
oaire.citation.startPage12pt_PT
oaire.citation.titleCortexpt_PT
oaire.citation.volume150pt_PT
person.familyNamede Mendonça
person.familyNameCastelo-Branco
person.familyNameMaruta
person.familyNameMiltenberger-Miltenyi
person.familyNameVerdelho
person.givenNameAlexandre
person.givenNameMiguel
person.givenNameCarolina
person.givenNameGabriel
person.givenNameAna
person.identifier351267
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person.identifier.scopus-author-id6701423628
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rcaap.rightsopenAccesspt_PT
rcaap.typearticlept_PT
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