Voluntary Running Improves Behavioral and Structural Abnormalities in a Mouse Model of CDKL5 Deficiency Disorder DOI Open Access
Nicola Mottolese, Beatrice Uguagliati, Marianna Tassinari

и другие.

Опубликована: Сен. 7, 2023

Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a rare neurodevelopmental disease caused by mutations in the X-linked CDKL5 gene. CDD characterized broad spectrum of clinical manifestations, including early-onset refractory epileptic seizures, intellectual disability, hypotonia, visual disturbances, and autism-like features. The Cdkl5 knockout (KO) mouse recapitulates several features CDD, autistic-like behavior, impaired learning memory, motor stereotypies. These behavioral alterations are accompanied diminished neuronal maturation survival, reduced dendritic branching spine maturation, marked microglia activation. There currently no cure or effective treatment to ameliorate symptoms disease. Aerobic exercise known exert multiple beneficial effects brain, not only increasing neurogenesis, but also improving cognitive tasks. To date, studies have analyzed effect physical on phenotype model. In view positive voluntary running brain models various human disorders, we sought determine whether daily running, sustained over month, could improve development defects KO mice. Our study showed that long-term improved hyperlocomotion impulsivity behaviors, memory performance This correlated with increased hippocampal inhibition structural improvements were associated BDNF levels. Given function, present findings support benefits as an adjuvant therapy for CDD.

Язык: Английский

Voluntary Running Improves Behavioral and Structural Abnormalities in a Mouse Model of CDKL5 Deficiency Disorder DOI Open Access
Nicola Mottolese, Beatrice Uguagliati, Marianna Tassinari

и другие.

Опубликована: Сен. 7, 2023

Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a rare neurodevelopmental disease caused by mutations in the X-linked CDKL5 gene. CDD characterized broad spectrum of clinical manifestations, including early-onset refractory epileptic seizures, intellectual disability, hypotonia, visual disturbances, and autism-like features. The Cdkl5 knockout (KO) mouse recapitulates several features CDD, autistic-like behavior, impaired learning memory, motor stereotypies. These behavioral alterations are accompanied diminished neuronal maturation survival, reduced dendritic branching spine maturation, marked microglia activation. There currently no cure or effective treatment to ameliorate symptoms disease. Aerobic exercise known exert multiple beneficial effects brain, not only increasing neurogenesis, but also improving cognitive tasks. To date, studies have analyzed effect physical on phenotype model. In view positive voluntary running brain models various human disorders, we sought determine whether daily running, sustained over month, could improve development defects KO mice. Our study showed that long-term improved hyperlocomotion impulsivity behaviors, memory performance This correlated with increased hippocampal inhibition structural improvements were associated BDNF levels. Given function, present findings support benefits as an adjuvant therapy for CDD.

Язык: Английский

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