Article
Human mutations in high-confidence Tourette disorder genes affect sensorimotor behavior, reward learning, and striatal dopamine in mice.
Proceedings of the National Academy of Sciences of the United States of America - 7 May 2024
Nasello Cara, Poppi Lauren A, Wu Junbing, Kowalski Tess F, Thackray Joshua K, Wang Riley, Persaud Angelina, Mahboob Mariam, Lin Sherry, Spaseska Rodna, Johnson C K, Gordon Derek, Tissir Fadel, Heiman Gary A, Tischfield Jay A, Bocarsly Miriam, Tischfield Max A
Abstract excerpt
Tourette disorder (TD) is poorly understood, despite affecting 1/160 children. A lack of animal models possessing construct, face, and predictive validity hinders progress in the field. We used CRISPR/Cas9 genome editing to generate mice with mutations orthologous to human de novo variants in two high-confidence Tourette genes, CELSR3 and WWC1. Mice with human mutations in Celsr3 and Wwc1 exhibit cognitive and/or...
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