Neuropeptide release is impaired in a mouse model of fragile x mental retardation syndrome

Suresh P. Annangudi, Agatha E. Luszpak, Soong Ho Kim, Shifang Ren, Nathan G. Hatcher, Ivan Jeanne Weiler, Keith T. Thornley, Brian M. Kile, R. Mark Wightman, William T. Greenough, Jonathan V Sweedler

Research output: Contribution to journalArticle

Abstract

Fragile X syndrome (FXS), an inherited disorder characterized by mental retardation and autism-like behaviors, is caused by the failure to transcribe the gene for fragile X mental retardation protein (FMRP), a translational regulator and transporter of select mRNAs. FXS model mice (Fmr1 KO mice) exhibit impaired neuropeptide release. Release of biogenic amines does not differ between wild-type (WT) and Fmr1 KO mice. Rab3A, an mRNA cargo of FMRP involved in the recruitment of vesicles, is decreased by â50% in synaptoneurosomes of Fmr1 KO mice; however, the number of dense-core vesicles (DCVs) does not differ between WT and Fmr1 KO mice. Therefore, deficits associated with FXS may reflect this aberrant vesicle release, specifically involving docking and fusion of peptidergic DCVs, and may lead to defective maturation and maintenance of synaptic connections.

Original languageEnglish (US)
Pages (from-to)306-314
Number of pages9
JournalACS Chemical Neuroscience
Volume1
Issue number4
DOIs
StatePublished - Apr 21 2010

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Fragile X Mental Retardation Protein
Neuropeptides
Intellectual Disability
Fragile X Syndrome
Messenger RNA
Biogenic Amines
Secretory Vesicles
Fusion reactions
Genes
Autistic Disorder
Maintenance

ASJC Scopus subject areas

  • Biochemistry
  • Physiology
  • Cognitive Neuroscience
  • Cell Biology

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Neuropeptide release is impaired in a mouse model of fragile x mental retardation syndrome. / Annangudi, Suresh P.; Luszpak, Agatha E.; Kim, Soong Ho; Ren, Shifang; Hatcher, Nathan G.; Weiler, Ivan Jeanne; Thornley, Keith T.; Kile, Brian M.; Wightman, R. Mark; Greenough, William T.; Sweedler, Jonathan V.

In: ACS Chemical Neuroscience, Vol. 1, No. 4, 21.04.2010, p. 306-314.

Research output: Contribution to journalArticle

Annangudi, SP, Luszpak, AE, Kim, SH, Ren, S, Hatcher, NG, Weiler, IJ, Thornley, KT, Kile, BM, Wightman, RM, Greenough, WT & Sweedler, JV 2010, 'Neuropeptide release is impaired in a mouse model of fragile x mental retardation syndrome', ACS Chemical Neuroscience, vol. 1, no. 4, pp. 306-314. https://doi.org/10.1021/cn900036x
Annangudi SP, Luszpak AE, Kim SH, Ren S, Hatcher NG, Weiler IJ et al. Neuropeptide release is impaired in a mouse model of fragile x mental retardation syndrome. ACS Chemical Neuroscience. 2010 Apr 21;1(4):306-314. https://doi.org/10.1021/cn900036x
Annangudi, Suresh P. ; Luszpak, Agatha E. ; Kim, Soong Ho ; Ren, Shifang ; Hatcher, Nathan G. ; Weiler, Ivan Jeanne ; Thornley, Keith T. ; Kile, Brian M. ; Wightman, R. Mark ; Greenough, William T. ; Sweedler, Jonathan V. / Neuropeptide release is impaired in a mouse model of fragile x mental retardation syndrome. In: ACS Chemical Neuroscience. 2010 ; Vol. 1, No. 4. pp. 306-314.
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