Literature DB >> 22009360

Fragile X syndrome and targeted treatment trials.

Randi Hagerman1, Julie Lauterborn, Jacky Au, Elizabeth Berry-Kravis.   

Abstract

Work in recent years has revealed an abundance of possible new treatment targets for fragile X syndrome (FXS). The use of animal models, including the fragile X knockout mouse which manifests a phenotype very similar to FXS in humans, has resulted in great strides in this direction of research. The lack of Fragile X Mental Retardation Protein (FMRP) in FXS causes dysregulation and usually overexpression of a number of its target genes, which can cause imbalances of neurotransmission and deficits in synaptic plasticity. The use of metabotropic glutamate receptor (mGluR) blockers and gamma amino-butyric acid (GABA) agonists have been shown to be efficacious in reversing cellular and behavioral phenotypes, and restoring proper brain connectivity in the mouse and fly models. Proposed new pharmacological treatments and educational interventions are discussed in this chapter. In combination, these various targeted treatments show promising preliminary results in mitigating or even reversing the neurobiological abnormalities caused by loss of FMRP, with possible translational applications to other neurodevelopmental disorders including autism.

Entities:  

Mesh:

Substances:

Year:  2012        PMID: 22009360      PMCID: PMC4114775          DOI: 10.1007/978-3-642-21649-7_17

Source DB:  PubMed          Journal:  Results Probl Cell Differ        ISSN: 0080-1844


  226 in total

1.  Mildly impaired water maze performance in male Fmr1 knockout mice.

Authors:  R D'Hooge; G Nagels; F Franck; C E Bakker; E Reyniers; K Storm; R F Kooy; B A Oostra; P J Willems; P P De Deyn
Journal:  Neuroscience       Date:  1997-01       Impact factor: 3.590

2.  Behavioral and neuroanatomical characterization of the Fmr1 knockout mouse.

Authors:  Yann S Mineur; Frans Sluyter; Sanne de Wit; Ben A Oostra; Wim E Crusio
Journal:  Hippocampus       Date:  2002       Impact factor: 3.899

3.  Epilepsy and EEG findings in males with fragile X syndrome.

Authors:  S A Musumeci; R J Hagerman; R Ferri; P Bosco; B Dalla Bernardina; C A Tassinari; G B De Sarro; M Elia
Journal:  Epilepsia       Date:  1999-08       Impact factor: 5.864

4.  Metabotropic receptor-dependent long-term depression persists in the absence of protein synthesis in the mouse model of fragile X syndrome.

Authors:  Elena D Nosyreva; Kimberly M Huber
Journal:  J Neurophysiol       Date:  2006-02-01       Impact factor: 2.714

5.  Audiogenic seizure susceptibility is reduced in fragile X knockout mice after introduction of FMR1 transgenes.

Authors:  Sebastiano A Musumeci; Giuseppe Calabrese; Carmela M Bonaccorso; Simona D'Antoni; Judith R Brouwer; Cathy E Bakker; Maurizio Elia; Raffaele Ferri; David L Nelson; Ben A Oostra; Maria Vincenza Catania
Journal:  Exp Neurol       Date:  2006-09-27       Impact factor: 5.330

6.  Evidence for social anxiety and impaired social cognition in a mouse model of fragile X syndrome.

Authors:  Caitlyn H McNaughton; Jisook Moon; Myla S Strawderman; Kenneth N Maclean; Jeffrey Evans; Barbara J Strupp
Journal:  Behav Neurosci       Date:  2008-04       Impact factor: 1.912

Review 7.  Psychopharmacology in fragile X syndrome--present and future.

Authors:  Elizabeth Berry-Kravis; Kristina Potanos
Journal:  Ment Retard Dev Disabil Res Rev       Date:  2004

8.  Altered cortical synaptic morphology and impaired memory consolidation in forebrain- specific dominant-negative PAK transgenic mice.

Authors:  Mansuo L Hayashi; Se-Young Choi; B S Shankaranarayana Rao; Hae-Yoon Jung; Hey-Kyoung Lee; Dawei Zhang; Sumantra Chattarji; Alfredo Kirkwood; Susumu Tonegawa
Journal:  Neuron       Date:  2004-06-10       Impact factor: 17.173

9.  Extracellular signal-regulated protein kinase activation is required for metabotropic glutamate receptor-dependent long-term depression in hippocampal area CA1.

Authors:  Sean M Gallagher; Christine A Daly; Mark F Bear; Kimberly M Huber
Journal:  J Neurosci       Date:  2004-05-19       Impact factor: 6.167

10.  Positive AMPA receptor modulation rapidly stimulates BDNF release and increases dendritic mRNA translation.

Authors:  Hussam Jourdi; Yu-Tien Hsu; Miou Zhou; Qingyu Qin; Xiaoning Bi; Michel Baudry
Journal:  J Neurosci       Date:  2009-07-08       Impact factor: 6.167

View more
  50 in total

1.  Fragile X Syndrome FMRP Co-localizes with Regulatory Targets PSD-95, GABA Receptors, CaMKIIα, and mGluR5 at Fiber Cell Membranes in the Eye Lens.

Authors:  Peter H Frederikse; Anoop Nandanoor; Chinnaswamy Kasinathan
Journal:  Neurochem Res       Date:  2015-08-23       Impact factor: 3.996

2.  Translational regulation of NeuroD1 expression by FMRP: involvement in glutamatergic neuronal differentiation of cultured rat primary neural progenitor cells.

Authors:  Se Jin Jeon; Ji-Woon Kim; Ki Chan Kim; So Min Han; Hyo Sang Go; Jung Eun Seo; Chang Soon Choi; Jong Hoon Ryu; Chan Young Shin; Mi-Ryoung Song
Journal:  Cell Mol Neurobiol       Date:  2013-12-12       Impact factor: 5.046

3.  Autism Symptomatology in Boys with Fragile X Syndrome: A Cross Sectional Developmental Trajectories Comparison with Nonsyndromic Autism Spectrum Disorder.

Authors:  Angela John Thurman; Andrea McDuffie; Sara T Kover; Randi J Hagerman; Leonard Abbeduto
Journal:  J Autism Dev Disord       Date:  2015-09

4.  PDE-4 inhibition rescues aberrant synaptic plasticity in Drosophila and mouse models of fragile X syndrome.

Authors:  Catherine H Choi; Brian P Schoenfeld; Eliana D Weisz; Aaron J Bell; Daniel B Chambers; Joseph Hinchey; Richard J Choi; Paul Hinchey; Maria Kollaros; Michael J Gertner; Neal J Ferrick; Allison M Terlizzi; Nicole Yohn; Eric Koenigsberg; David A Liebelt; R Suzanne Zukin; Newton H Woo; Michael R Tranfaglia; Natalia Louneva; Steven E Arnold; Steven J Siegel; Francois V Bolduc; Thomas V McDonald; Thomas A Jongens; Sean M J McBride
Journal:  J Neurosci       Date:  2015-01-07       Impact factor: 6.167

5.  Prevalence and Predictors of Anxiety Disorders in Adolescent and Adult Males with Autism Spectrum Disorder and Fragile X Syndrome.

Authors:  Jordan Ezell; Abigail Hogan; Amanda Fairchild; Kimberly Hills; Jessica Klusek; Leonard Abbeduto; Jane Roberts
Journal:  J Autism Dev Disord       Date:  2019-03

6.  FRAGILE X SYNDROME: PSYCHIATRIC MANIFESTATIONS, ASSESSMENT AND EMERGING THERAPIES.

Authors:  Paula M Wadell; Randi J Hagerman; David R Hessl
Journal:  Curr Psychiatry Rev       Date:  2013-02-01

7.  Newborn screening and cascade testing for FMR1 mutations.

Authors:  Page L Sorensen; Louise W Gane; Mark Yarborough; Randi J Hagerman; Flora Tassone
Journal:  Am J Med Genet A       Date:  2012-12-13       Impact factor: 2.802

8.  Glycogen synthase kinase-3 inhibitors reverse deficits in long-term potentiation and cognition in fragile X mice.

Authors:  Aimee V Franklin; Margaret K King; Valle Palomo; Ana Martinez; Lori L McMahon; Richard S Jope
Journal:  Biol Psychiatry       Date:  2013-09-13       Impact factor: 13.382

9.  Impaired activity-dependent FMRP translation and enhanced mGluR-dependent LTD in Fragile X premutation mice.

Authors:  Adam J Iliff; Abigail J Renoux; Amy Krans; Karen Usdin; Michael A Sutton; Peter K Todd
Journal:  Hum Mol Genet       Date:  2012-12-18       Impact factor: 6.150

10.  A 'cure' for Down syndrome: what do parents want?

Authors:  A Inglis; Z Lohn; J C Austin; C Hippman
Journal:  Clin Genet       Date:  2014-03-21       Impact factor: 4.438

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.