Interplay of LIS1 and MeCP2: Interactions and Implications With the Neurodevelopmental Disorders Lissencephaly and Rett Syndrome

Liraz Keidar, Gabi Gerlitz, Aditya Kshirsagar, Michael Tsoory, Tsviya Olender, Xing Wang, Ying Yang, Yu Sheng Chen, Yun Gui Yang, Irina Voineagu, Orly Reiner

פרסום מחקרי: פרסום בכתב עתמאמרביקורת עמיתים

8 ציטוטים ‏(Scopus)

תקציר

LIS1 is the main causative gene for lissencephaly, while MeCP2 is the main causative gene for Rett syndrome, both of which are neurodevelopmental diseases. Here we report nuclear functions for LIS1 and identify previously unrecognized physical and genetic interactions between the products of these two genes in the cell nucleus, that has implications on MeCP2 organization, neuronal gene expression and mouse behavior. Reduced LIS1 levels affect the association of MeCP2 with chromatin. Transcriptome analysis of primary cortical neurons derived from wild type, Lis1±, MeCP2−/y, or double mutants mice revealed a large overlap in the differentially expressed (DE) genes between the various mutants. Overall, our findings provide insights on molecular mechanisms involved in the neurodevelopmental disorders lissencephaly and Rett syndrome caused by dysfunction of LIS1 and MeCP2, respectively.

שפה מקוריתאנגלית
מספר המאמר370
כתב עתFrontiers in Cellular Neuroscience
כרך13
מזהי עצם דיגיטלי (DOIs)
סטטוס פרסוםפורסם - 14 אוג׳ 2019
פורסם באופן חיצוניכן

טביעת אצבע

להלן מוצגים תחומי המחקר של הפרסום 'Interplay of LIS1 and MeCP2: Interactions and Implications With the Neurodevelopmental Disorders Lissencephaly and Rett Syndrome'. יחד הם יוצרים טביעת אצבע ייחודית.

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