Dumontier, Dimitri, Liebman, Samuel A, Le, Viet-Hang, George, Shanu, Valdemar, Deasia, Van Aelst, Linda, Pouchelon, Gabrielle (September 2024) Restoring transient connectivity during development improves dysfunctions in fragile X mice. bioRxiv. ISSN 2692-8205 (Submitted)
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10.1101.2024.09.08.611918.pdf - Submitted Version Available under License Creative Commons Attribution Non-commercial No Derivatives. Download (3MB) | Preview |
Abstract
Early-generated circuits are critical for the maturation of cortical network activity and the formation of excitation/inhibition (E/I) balance. This process involves the maturation of specific populations of inhibitory neurons. While parvalbumin (PV)-expressing neurons have been associated with E/I impairments observed in neurodevelopmental disorders, somatostatin-expressing (SST) neurons have recently been shown to regulate PV neuron maturation by controlling neural dynamics in the developing cortex. SST neurons receive transient connections from the sensory thalamus, yet the implications of transient connectivity in neurodevelopmental disorders remain unknown. Here, we show that thalamocortical connectivity to SST neurons is persistent rather than transient in a mouse model of Fragile X syndrome. We were able to restore the transient dynamics using chemogenetics, which led to the recovery of fragile X-associated dysfunctions in circuit maturation and sensory-dependent behavior. Overall, our findings unveil the role of early transient dynamics in controlling downstream maturation of sensory functions.
| Item Type: | Paper |
|---|---|
| Subjects: | diseases & disorders > congenital hereditary genetic diseases diseases & disorders organism description > animal diseases & disorders > congenital hereditary genetic diseases > fragile X syndrome organism description > animal > mammal organism description > animal > mammal > rodent > mouse organism description > animal > mammal > rodent |
| CSHL Authors: | |
| Communities: | CSHL labs > Van Aelst lab CSHL labs > Pouchelon lab |
| SWORD Depositor: | CSHL Elements |
| Depositing User: | CSHL Elements |
| Date: | 9 September 2024 |
| Date Deposited: | 20 Sep 2024 17:44 |
| Last Modified: | 20 Sep 2024 17:44 |
| Related URLs: | |
| URI: | https://repository.cshl.edu/id/eprint/41674 |
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