دورية أكاديمية
أعرض في EDS

Adult Camk2a gene reinstatement restores the learning and plasticity deficits of Camk2a knockout mice.

التفاصيل البيبلوغرافية
العنوان: Adult Camk2a gene reinstatement restores the learning and plasticity deficits of Camk2a knockout mice.
المؤلفون: Rigter PMF; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Wallaard I; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Aghadavoud Jolfaei M; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Kingma J; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Post L; Department of Neuroscience, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Elgersma M; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., Elgersma Y; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands., van Woerden GM; Department of Clinical Genetics, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.; Department of Neuroscience, Erasmus Medical Center, 3015GD Rotterdam, the Netherlands.
المصدر: IScience [iScience] 2022 Oct 08; Vol. 25 (11), pp. 105303. Date of Electronic Publication: 2022 Oct 08 (Print Publication: 2022).
نوع المنشور: Journal Article
اللغة: English
بيانات الدورية: Publisher: Cell Press Country of Publication: United States NLM ID: 101724038 Publication Model: eCollection Cited Medium: Internet ISSN: 2589-0042 (Electronic) Linking ISSN: 25890042 NLM ISO Abbreviation: iScience Subsets: PubMed not MEDLINE
أسماء مطبوعة: Original Publication: [Cambridge, MA] : Cell Press, [2018]-
مستخلص: With the recent findings that mutations in the gene encoding the α-subunit of calcium/calmodulin-dependent protein kinase II (CAMK2A) causes a neurodevelopmental disorder (NDD), it is of great therapeutic relevance to know if there exists a critical developmental time window in which CAMK2A needs to be expressed for normal brain development, or whether expression of the protein at later stages is still beneficial to restore normal functioning. To answer this question, we generated an inducible Camk2a mouse model, which allows us to express CAMK2A at any desired time. Here, we show that adult expression of CAMK2A rescues the behavioral and electrophysiological phenotypes seen in the Camk2a knock-out mice, including spatial and conditional learning and synaptic plasticity. These results suggest that CAMK2A does not play a critical irreversible role in neurodevelopment, which is of importance for future therapies to treat CAMK2A-dependent disorders.
Competing Interests: The authors declare no competing interests.
(© 2022 The Author(s).)
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فهرسة مساهمة: Keywords: Cell biology; Cellular neuroscience; Developmental biology
تواريخ الأحداث: Date Created: 20221028 Latest Revision: 20221029
رمز التحديث: 20240628
مُعرف محوري في PubMed: PMC9593899
DOI: 10.1016/j.isci.2022.105303
PMID: 36304100
قاعدة البيانات: MEDLINE
الوصف
تدمد:2589-0042
DOI:10.1016/j.isci.2022.105303