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 journalArticle

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 languageEnglish
Pages (from-to)2581-2593.e5
JournalCell Reports
Volume28
Issue number10
DOIs
StatePublished - Sep 3 2019

Keywords

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

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