Wnd/DLK Is a Critical Target of FMRP Responsible for Neurodevelopmental and Behavior Defects in the Drosophila Model of Fragile X Syndrome

Alexandra Russo; Aaron DiAntonio

doi:10.1016/j.celrep.2019.08.001

Wnd/DLK Is a Critical Target of FMRP Responsible for Neurodevelopmental and Behavior Defects in the Drosophila Model of Fragile X Syndrome

Alexandra Russo, Aaron DiAntonio

Research output: Contribution to journal › Article › peer-review

7 Scopus citations

Abstract

Russo and DiAntonio identify a dysregulated MAPK signaling pathway in the fly model of fragile X syndrome. MAP3K Wnd/DLK drives dFmr1 mutant phenotypes, and pharmacological inhibition of Wnd/DLK prevents neural dysfunction in this model, thus highlighting a possible role for Wnd/DLK in the pathophysiology of fragile X syndrome.

Original language	English
Pages (from-to)	2581-2593.e5
Journal	Cell Reports
Volume	28
Issue number	10
DOIs	https://doi.org/10.1016/j.celrep.2019.08.001
State	Published - Sep 3 2019

Keywords

GNE-3511
JNK
grooming behavior
highwire
neurodegeneration
neurodevelopment
neuromuscular junction
synaptic transmission

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10.1016/j.celrep.2019.08.001

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title = "Wnd/DLK Is a Critical Target of FMRP Responsible for Neurodevelopmental and Behavior Defects in the Drosophila Model of Fragile X Syndrome",

abstract = "Russo and DiAntonio identify a dysregulated MAPK signaling pathway in the fly model of fragile X syndrome. MAP3K Wnd/DLK drives dFmr1 mutant phenotypes, and pharmacological inhibition of Wnd/DLK prevents neural dysfunction in this model, thus highlighting a possible role for Wnd/DLK in the pathophysiology of fragile X syndrome.",

keywords = "GNE-3511, JNK, grooming behavior, highwire, neurodegeneration, neurodevelopment, neuromuscular junction, synaptic transmission",

author = "Alexandra Russo and Aaron DiAntonio",

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AU - Russo, Alexandra

AU - DiAntonio, Aaron

PY - 2019/9/3

Y1 - 2019/9/3

N2 - Russo and DiAntonio identify a dysregulated MAPK signaling pathway in the fly model of fragile X syndrome. MAP3K Wnd/DLK drives dFmr1 mutant phenotypes, and pharmacological inhibition of Wnd/DLK prevents neural dysfunction in this model, thus highlighting a possible role for Wnd/DLK in the pathophysiology of fragile X syndrome.

AB - Russo and DiAntonio identify a dysregulated MAPK signaling pathway in the fly model of fragile X syndrome. MAP3K Wnd/DLK drives dFmr1 mutant phenotypes, and pharmacological inhibition of Wnd/DLK prevents neural dysfunction in this model, thus highlighting a possible role for Wnd/DLK in the pathophysiology of fragile X syndrome.

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KW - JNK

KW - grooming behavior

KW - highwire

KW - neurodegeneration

KW - neurodevelopment

KW - neuromuscular junction

KW - synaptic transmission

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ER -

Wnd/DLK Is a Critical Target of FMRP Responsible for Neurodevelopmental and Behavior Defects in the Drosophila Model of Fragile X Syndrome

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Keywords

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