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DOI 10.1093/brain/awac446

Медицинская библиотека · Brain : a journal of neurology · 2023

Neurodevelopmental effects of genetic frontotemporal dementia in young adult mutation carriers.

Finger Elizabeth, Malik Rubina, Bocchetta Martina, Coleman Kristy, Graff Caroline, Borroni Barbara, Masellis Mario, Laforce Robert, Greaves Caroline V, Russell Lucy L, Convery Rhian S, Bouzigues Arabella, Cash David M, Otto Markus, Synofzik Matthis, Rowe James B, Galimberti Daniela, Tiraboschi Pietro, Bartha Robert, Shoesmith Christen, Tartaglia Maria Carmela, van Swieten John C, Seelaar Harro, Jiskoot Lize C, Sorbi Sandro, Butler Chris R, Gerhard Alexander, Sanchez-Valle Raquel, de Mendonça Alexandre, Moreno Fermin, Vandenberghe Rik, Le Ber Isabelle, Levin Johannes, Pasquier Florence, Santana Isabel, Rohrer Jonathan D, Ducharme Simon, Genetic FTD Initiative, GENFI

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Аннотация

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.

Для цитирования: Finger Elizabeth, Malik Rubina, Bocchetta Martina, Coleman Kristy, Graff Caroline, Borroni Barbara, Masellis Mario, Laforce Robert, Greaves Caroline V, Russell Lucy L, Convery Rhian S, Bouzigues Arabella, Cash David M, Otto Markus, Synofzik Matthis, Rowe James B, Galimberti Daniela, Tiraboschi Pietro, Bartha Robert, Shoesmith Christen, Tartaglia Maria Carmela, van Swieten John C, Seelaar Harro, Jiskoot Lize C, Sorbi Sandro, Butler Chris R, Gerhard Alexander, Sanchez-Valle Raquel, de Mendonça Alexandre, Moreno Fermin, Vandenberghe Rik, Le Ber Isabelle, Levin Johannes, Pasquier Florence, Santana Isabel, Rohrer Jonathan D, Ducharme Simon, Genetic FTD Initiative, GENFI. Neurodevelopmental effects of genetic frontotemporal dementia in young adult mutation carriers.. Brain : a journal of neurology. 2023. DOI: 10.1093/brain/awac446.

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