Cytoplasmic FUS triggers early behavioral alterations linked to cortical neuronal hyperactivity and inhibitory synaptic defects.

doi:10.1038/s41467-021-23187-9

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Article

Cytoplasmic FUS triggers early behavioral alterations linked to cortical neuronal hyperactivity and inhibitory synaptic defects.

Nature communications - 21 May 2021

Scekic-Zahirovic Jelena, Sanjuan-Ruiz Inmaculada, Kan Vanessa, Megat Salim, De Rossi Pierre, Dieterlé Stéphane, Cassel Raphaelle, Jamet Marguerite, Kessler Pascal, Wiesner Diana, Tzeplaeff Laura, Demais Valérie, Sahadevan Sonu, Hembach Katharina M, Muller Hans-Peter, Picchiarelli Gina, Mishra Nibha, Antonucci Stefano, Dirrig-Grosch Sylvie, Kassubek Jan, Rasche Volker, Ludolph Albert, Boutillier Anne-Laurence, Roselli Francesco, Polymenidou Magdalini, Lagier-Tourenne Clotilde, Liebscher Sabine, Dupuis Luc

Abstract excerpt

Gene mutations causing cytoplasmic mislocalization of the RNA-binding protein FUS lead to severe forms of amyotrophic lateral sclerosis (ALS). Cytoplasmic accumulation of FUS is also observed in other diseases, with unknown consequences. Here, we show that cytoplasmic mislocalization of FUS drives behavioral abnormalities in knock-in mice, including locomotor hyperactivity and alterations in social interactions,...

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Identifiers

PMID: 34021132
DOI: 10.1038/s41467-021-23187-9
PMCID: PMC8140148

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