Intellectual Disability and Behavioral Deficits Linked to CYFIP1 Missense Variants Disrupting Actin Polymerization

Mariano, Vittoria; Kanellopoulos, Alexandros K.; Ricci, Carlotta; Di Marino, Daniele; Borrie, Sarah C.; Dupraz, Sebastian; Bradke, Frank; Achsel, Tilmann; Legius, Eric; Odent, Sylvie; Billuart, Pierre; Bienvenu, Thierry; Bagni, Claudia

doi:10.1016/j.biopsych.2023.08.027

Cited by 4 publications

(2 citation statements)

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“…Future research in the field will need to combine these various data to offer an integrated and more comprehensive understanding of how the players underlying social spacing behaviour work together. For example, there might be a structural association between CYF1P and Nlgs, as both affect the actin cytoskeleton [56,[101][102][103][104].…”

Section: Discussionmentioning

confidence: 99%

“…In addition to social isolation, social space is modulated by social experience [41]; physiological state such as hunger [46], age of the individual and age of the parents [46,47]; time of the day [46,48]; viral infections, as well as exposure to environmental toxins [49][50][51][52][53] or anti-oxidant [49]. Orthologues of several candidate genes for neurodevelopmental disorders in humans also influence social space in Drosophila, as reported for example, not exhaustively, in [54][55][56][57][58][59][60][61]. Finally, a number of sensory modalities, neurocircuitry and synaptic proteins underlying social spacing have been identified [42,43,[46][47][48][49]54,[61][62][63][64][65][66][67][68].…”

Section: Introductionmentioning

confidence: 99%

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Recovery from social isolation requires dopamine in males, but not the autism-related gene nlg3 in either sex

Yost,

Scott,

Kurbaj

et al. 2024

R. Soc. Open Sci.

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Social isolation causes profound changes in social behaviour in a variety of species. However, the genetic and molecular mechanisms modulating behavioural responses to social isolation and social recovery remain to be elucidated. Here, we quantified the behavioural response of vinegar flies to social isolation using two distinct protocols (social space preference and sociability, the spontaneous tendencies to form groups). We found that social isolation increased social space and reduced sociability. These effects of social isolation were reversible and could be reduced after 3 days of group housing. Flies with a loss of function of neuroligin3 (orthologue of autism-related neuroligin genes) with known increased social space in a socially enriched environment were still able to recover from social isolation. We also show that dopamine (DA) is needed for a response to social isolation and recovery in males but not in females. Furthermore, only in males, DA levels are reduced after isolation and are not recovered after group housing. Finally, in socially enriched flies mutant for neuroligin3 , DA levels are reduced in males, but not in females. We propose a model to explain how DA and neuroligin3 are involved in the behavioural response to social isolation and its recovery in a dynamic and sex-specific manner.

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