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• W4280633927 abstract "Abstract Rett syndrome is characterized by an early period of typical development and then, regression of learned motor and speech skills in girls. Loss of MECP2 protein is thought to cause Rett syndrome phenotypes. The specific underlying mechanisms from typical developmental trajectory to regression features throughout life are unclear. Lack of established timelines to study the molecular, cellular, and behavioral features of regression in female mouse models is a major contributing factor. Due to random X-chromosome inactivation, female patients with Rett syndrome and female mouse models for Rett syndrome ( Mecp2 Heterozygous , Het) express a functional copy of wild-type MECP2 protein in approximately half of all cells. As MECP2 expression is regulated during early postnatal development and experience, we characterized the expression of wild-type MECP2 in the primary somatosensory cortex of female Het mice. Here, we report increased MECP2 levels in non-parvalbumin-positive neurons of 6-week-old adolescent Het relative to age-matched wild-type controls, while also displaying similar levels of perineuronal net expression, mild tactile sensory perception deficits, and efficient pup retrieval behavior. In contrast, 12-week-old adult Het express MECP2 at levels similar to age-matched wild-type mice, show increased perineuronal net expression in the cortex, and display significant tactile sensory perception deficits. Thus, we have identified a set of behavioral metrics and the cellular substrates to study regression during a specific time in the female Het mouse model, which coincide with changes in wild-type MECP2 expression. We speculate that the precocious increase in MECP2 expression within specific cell types of adolescent Het may provide compensatory benefits at the behavioral level, while the inability to further increase MECP2 levels leads to regressive behavioral phenotypes over time." @default.
• W4280633927 created "2022-05-22" @default.
• W4280633927 creator A5024051950 @default.
• W4280633927 creator A5033685076 @default.
• W4280633927 creator A5038631781 @default.
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• W4280633927 creator A5050699822 @default.
• W4280633927 creator A5069870553 @default.
• W4280633927 creator A5088932134 @default.
• W4280633927 date "2022-04-25" @default.
• W4280633927 modified "2023-09-26" @default.
• W4280633927 title "Wild-type MECP2 expression coincides with age-dependent sensory phenotypes in a female mouse model for Rett syndrome" @default.
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