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CELSR3 deficiency leads to tic-related behaviors and dopaminergic alterations in the striatum

GSE249902 Mus musculus Expression profiling by high throughput sequencing 6 samples Submitted 2025/06/12 Platform GPL24247
Summary
The gene CELSR3 (cadherin EGF LAG seven‐pass-G‐type receptor 3) has been recently recognized as a high-confidence risk factor for tic disorders (TD). Celsr3 mutant mice displayed tic-like grooming stereotypies and jerks, as well as sensorimotor gating deficits, which were opposed by TD therapies. Single-nucleus transcriptomic analyses revealed that Celsr3 mutants featured a unique group of eccentric striatal projection neurons. Notably, the Drd3 gene, encoding the dopamine D3 receptor, was significantly upregulated in these cells as well as striosomal D1-positive neurons, while it was reduced in calretinin-positive GABAergic interneurons.
Published in
Tic-related behaviors in Celsr3 mutant mice are contributed by alterations of striatal D(3) dopamine receptors
Cadeddu R, Branca C, Braccagni G et al. · Molecular psychiatry 2025 · PMID 40155412 · doi:10.1038/s41380-025-02970-w
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Also filed as BioProject PRJNA1051112 and SRA study SRP477478. Searching any of these in the dataset finder brings you back here.

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