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@ARTICLE{Finger:169169,
author = {Finger, Elizabeth and Malik, Rubina and Bocchetta, Martina
and Coleman, Kristy and Graff, Caroline and Borroni, Barbara
and Masellis, Mario and Laforce, Robert and Greaves,
Caroline V and Russell, Lucy L and Convery, Rhian S and
Bouzigues, Arabella and Cash, David M and Otto, Markus and
Synofzik, Matthis and Rowe, James B and Galimberti, Daniela
and Tiraboschi, Pietro and Bartha, Robert and Shoesmith,
Christen and Tartaglia, Maria Carmela and van Swieten, John
C and Seelaar, Harro and Jiskoo, Lize C and Sorbi, Sandro
and Butler, Chris R and Gerhard, Alexander and
Sanchez-Valle, Raquel and de Mendonça, Alexandre and
Moreno, Fermin and Vandenberghe, Rik and Le Ber, Isabelle
and Levin, Johannes and Pasquier, Florence and Santana,
Isabel and Rohrer, Jonathan D and Ducharme, Simon},
collaboration = {GENFI, Genetic FTD Initiative},
othercontributors = {Esteve, Aitana Sogorb and Heller, Carolin and Thomas, David
L and Todd, Emily G and Nicholas, Jennifer and Benotmane,
Hanya and Zetterberg, Henrik and Swift, Imogen J and Samra,
Kiran and Shafei, Rachelle and Timberlake, Carolyn and Cope,
Thomas and Rittman, Timothy and Benussi, Alberto and Premi,
Enrico and Gasparotti, Roberto and Archetti, Silvana and
Gazzina, Stefano and Cantoni, Valentina and Arighi, Andrea
and Fenoglio, Chiara and Scarpini, Elio and Fumagalli,
Giorgio and Borracci, Vittoria and Rossi, Giacomina and
Giaccone, Giorgio and Di Fede, Giuseppe and Caroppo, Paola
and Tiraboschi, Pietro and Prioni, Sara and Redaelli,
Veronica and Tang-Wai, David and Rogaeva, Ekaterina and
Castelo-Branco, Miguel and Freedman, Morris and Keren, Ron
and Black, Sandra and Mitchell, Sara and Rademakers, Rosa
and Poos, Jackie and Papma, Janne M and Giannini, Lucia and
van Minkelen, Rick and Pijnenburg, Yolande and Nacmias,
Benedetta and Ferrari, Camilla and Polito, Cristina and
Lombardi, Gemma and Bessi, Valentina and Veldsman, Michele
and Andersson, Christin and Thonberg, Hakan and Öijerstedt,
Linn and Jelic, Vesna and Thompson, Paul and Langheinrich,
Tobias and Lladó, Albert and Antonell, Anna and Olives,
Jaume and Balasa, Mircea and Bargalló, Nuria and
Borrego-Ecija, Sergi and Verdelho, Ana and Maruta, Carolina
and Ferreira, Catarina B and Miltenberger, Gabriel and do
Couto, Frederico Simões and Gabilondo, Alazne and
Gorostidi, Ana and Villanua, Jorge and Cañada, Marta and
Tainta, Mikel and Zulaica, Miren and Barandiaran, Myriam and
Alves, Patricia and Bender, Benjamin and Wilke, Carlo and
Graf, Lisa and Vogels, Annick and Vandenbulcke, Mathieu and
Van Damme, Philip and Bruffaerts, Rose and Poesen, Koen and
Rosa-Neto, Pedro and Gauthier, Serge and Camuzat, Agnès and
Brice, Alexis and Bertrand, Anne and Funkiewiez, Aurélie
and Rinaldi, Daisy and Saracino, Dario and Colliot, Olivier
and Sayah, Sabrina and Prix, Catharina and Wlasich,
Elisabeth and Wagemann, Olivia and Loosli, Sandra and
Schönecker, Sonja and Hoegen, Tobias and Lombardi, Jolina
and Anderl-Straub, Sarah and Rollin, Adeline and Kuchcinski,
Gregory and Bertoux, Maxime and Lebouvier, Thibaud and
Deramecourt, Vincent and Santiago, Beatriz and Duro, Diana
and Leitão, Maria João and Almeida, Maria Rosario and
Tábuas-Pereira, Miguel and Afonso, Sónia},
title = {{N}eurodevelopmental effects of genetic frontotemporal
dementia in young adult mutation carriers},
journal = {Brain},
volume = {146},
number = {5},
issn = {0006-8950},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {DZNE-2023-00048},
pages = {2120-2131},
year = {2023},
abstract = {While frontotemporal dementia has been considered a
neurodegenerative disease that starts in mid-life or later,
it is now clearly established that cortical and subcortical
volume loss is observed more than a decade prior to symptom
onset and progresses with ageing. To test the hypothesis
that genetic mutations causing frontotemporal dementia have
neurodevelopmental consequences, we examined the youngest
adults in the GENFI cohort of pre-symptomatic frontotemporal
dementia mutation carriers who are between 19 and 30 years
of age. Structural brain differences and improved
performance on some cognitive tests were found for MAPT and
GRN mutation carriers relative to familial non-carriers,
while smaller volumes were observed in C9orf72 repeat
expansion carriers at a mean age of 26 years. The detection
of such early differences supports potential advantageous
neurodevelopmental consequences of some frontotemporal
dementia-causing genetic mutations. These results have
implications for the design of therapeutic interventions for
frontotemporal dementia. Future studies at younger ages are
needed to identify specific early pathophysiologic or
compensatory processes that occur during the
neurodevelopmental period.},
keywords = {Humans / Young Adult / Adult / Frontotemporal Dementia:
genetics / Neurodegenerative Diseases / Progranulins:
genetics / Brain / Pick Disease of the Brain / Mutation /
C9orf72 Protein: genetics / tau Proteins: genetics /
Progranulins (NLM Chemicals) / C9orf72 (Other) / GRN (Other)
/ MAPT (Other) / frontotemporal dementia (Other) /
neurodevelopment (Other) / C9orf72 Protein (NLM Chemicals) /
tau Proteins (NLM Chemicals)},
cin = {AG Gasser 1 / Clinical Dementia Research München ; AG
Levin},
ddc = {610},
cid = {I:(DE-2719)1210000 / I:(DE-2719)1111016},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:36458975},
doi = {10.1093/brain/awac446},
url = {https://pub.dzne.de/record/169169},
}