Literature DB >> 24348096

Recent Progress in Rett Syndrome and MeCP2 Dysfunction: Assessment of Potential Treatment Options.

Christopher A Chapleau1, Jane Lane2, Jennifer Larimore3, Wei Li1, Lucas Pozzo-Miller1, Alan K Percy4.   

Abstract

Synaptic communication is highly regulated process of contact between cells allowing information to be stored and modified. Synaptic formation and maturation is the result of interactions between intrinsic genetic/molecular factors and the external environment to establish the communication in the brain. One disorder associated with faulty synapse communication is Rett Syndrome (RTT). RTT is the leading form of severe MR in females, affecting approximately 1:10,000 females worldwide, without predisposition to any particular racial or ethnic group. Mutations in MECP2, the gene encoding methyl-CpG-binding protein-2, have been identified in more than 95% of individuals with RTT. Birth and the milestones of early development appear to be normal in individuals with RTT until approximately 6-18 months when in the subsequent months and years that follows, physical, motor, and social-cognitive development enter a period of regression. The clinical management of these individuals is extremely multifaceted, relying on collaborations of specialists and researchers from many different fields. In this critical literature review, we provide an overview of Rett Syndrome, from patient to pathophysiology with a therapeutic summary of clinical trials in RTT and preclinical studies using mouse and cell models of RTT.

Entities:  

Keywords:  Clinical Trials; MeCP2; Neurodevelopmental Disorders; Rett Syndrome

Year:  2013        PMID: 24348096      PMCID: PMC3859379          DOI: 10.2217/fnl.12.79

Source DB:  PubMed          Journal:  Future Neurol        ISSN: 1479-6708


  32 in total

1.  Dendritic spine pathologies in hippocampal pyramidal neurons from Rett syndrome brain and after expression of Rett-associated MECP2 mutations.

Authors:  Christopher A Chapleau; Gaston D Calfa; Meredith C Lane; Asher J Albertson; Jennifer L Larimore; Shinichi Kudo; Dawna L Armstrong; Alan K Percy; Lucas Pozzo-Miller
Journal:  Neurobiol Dis       Date:  2009-05-12       Impact factor: 5.996

2.  Selective dendritic alterations in the cortex of Rett syndrome.

Authors:  D Armstrong; J K Dunn; B Antalffy; R Trivedi
Journal:  J Neuropathol Exp Neurol       Date:  1995-03       Impact factor: 3.685

3.  The disease progression of Mecp2 mutant mice is affected by the level of BDNF expression.

Authors:  Qiang Chang; Gargi Khare; Vardhan Dani; Sacha Nelson; Rudolf Jaenisch
Journal:  Neuron       Date:  2006-02-02       Impact factor: 17.173

4.  Longevity in Rett syndrome: analysis of the North American Database.

Authors:  Russell S Kirby; Jane B Lane; Jerry Childers; Steve A Skinner; Fran Annese; Judy O Barrish; Daniel G Glaze; Patrick Macleod; Alan K Percy
Journal:  J Pediatr       Date:  2010-01       Impact factor: 4.406

5.  Bdnf overexpression in hippocampal neurons prevents dendritic atrophy caused by Rett-associated MECP2 mutations.

Authors:  Jennifer L Larimore; Christopher A Chapleau; Shinichi Kudo; Anne Theibert; Alan K Percy; Lucas Pozzo-Miller
Journal:  Neurobiol Dis       Date:  2009-01-03       Impact factor: 5.996

Review 6.  Pharmaceuticals targeting nonsense mutations in genetic diseases: progress in development.

Authors:  Steven M Rowe; John P Clancy
Journal:  BioDrugs       Date:  2009       Impact factor: 5.807

7.  Partial reversal of Rett Syndrome-like symptoms in MeCP2 mutant mice.

Authors:  Daniela Tropea; Emanuela Giacometti; Nathan R Wilson; Caroline Beard; Cortina McCurry; Dong Dong Fu; Ruth Flannery; Rudolf Jaenisch; Mriganka Sur
Journal:  Proc Natl Acad Sci U S A       Date:  2009-02-10       Impact factor: 11.205

8.  Genetic and physical mapping of a gene encoding a methyl CpG binding protein, Mecp2, to the mouse X chromosome.

Authors:  N A Quaderi; R R Meehan; P H Tate; S H Cross; A P Bird; A Chatterjee; G E Herman; S D Brown
Journal:  Genomics       Date:  1994-08       Impact factor: 5.736

9.  Rett syndrome mutation MeCP2 T158A disrupts DNA binding, protein stability and ERP responses.

Authors:  Darren Goffin; Megan Allen; Le Zhang; Maria Amorim; I-Ting Judy Wang; Arith-Ruth S Reyes; Amy Mercado-Berton; Caroline Ong; Sonia Cohen; Linda Hu; Julie A Blendy; Gregory C Carlson; Steve J Siegel; Michael E Greenberg; Zhaolan Zhou
Journal:  Nat Neurosci       Date:  2011-11-27       Impact factor: 24.884

10.  MeCP2, a key contributor to neurological disease, activates and represses transcription.

Authors:  Maria Chahrour; Sung Yun Jung; Chad Shaw; Xiaobo Zhou; Stephen T C Wong; Jun Qin; Huda Y Zoghbi
Journal:  Science       Date:  2008-05-30       Impact factor: 47.728

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  13 in total

1.  β2-Adrenergic receptor agonist ameliorates phenotypes and corrects microRNA-mediated IGF1 deficits in a mouse model of Rett syndrome.

Authors:  Nikolaos Mellios; Jonathan Woodson; Rodrigo I Garcia; Benjamin Crawford; Jitendra Sharma; Steven D Sheridan; Stephen J Haggarty; Mriganka Sur
Journal:  Proc Natl Acad Sci U S A       Date:  2014-06-23       Impact factor: 11.205

Review 2.  Rett Syndrome: Reaching for Clinical Trials.

Authors:  Lucas Pozzo-Miller; Sandipan Pati; Alan K Percy
Journal:  Neurotherapeutics       Date:  2015-07       Impact factor: 7.620

3.  An RNA interference screen identifies druggable regulators of MeCP2 stability.

Authors:  Laura M Lombardi; Manar Zaghlula; Yehezkel Sztainberg; Steven A Baker; Tiemo J Klisch; Amy A Tang; Eric J Huang; Huda Y Zoghbi
Journal:  Sci Transl Med       Date:  2017-08-23       Impact factor: 17.956

4.  Illness Severity, Social and Cognitive Ability, and EEG Analysis of Ten Patients with Rett Syndrome Treated with Mecasermin (Recombinant Human IGF-1).

Authors:  Giorgio Pini; Laura Congiu; Alberto Benincasa; Pietro DiMarco; Stefania Bigoni; Adam H Dyer; Niall Mortimer; Andrea Della-Chiesa; Sean O'Leary; Rachel McNamara; Kevin J Mitchell; Michael Gill; Daniela Tropea
Journal:  Autism Res Treat       Date:  2016-01-26

5.  Improved MECP2 Gene Therapy Extends the Survival of MeCP2-Null Mice without Apparent Toxicity after Intracisternal Delivery.

Authors:  Sarah E Sinnett; Ralph D Hector; Kamal K E Gadalla; Clifford Heindel; Daphne Chen; Violeta Zaric; Mark E S Bailey; Stuart R Cobb; Steven J Gray
Journal:  Mol Ther Methods Clin Dev       Date:  2017-04-19       Impact factor: 6.698

6.  An early seizure variant type of a male Rett syndrome patient with a MECP2 p.Arg133His missense mutation.

Authors:  Jin A Yoon; Yongjin Yoo; Je Sang Lee; Young Mi Kim; Yong Beom Shin
Journal:  Mol Genet Genomic Med       Date:  2018-12-19       Impact factor: 2.183

7.  Potent hERG channel inhibition by sarizotan, an investigative treatment for Rett Syndrome.

Authors:  Hongwei Cheng; Chunyun Du; Yihong Zhang; Andrew F James; Christopher E Dempsey; Ana P Abdala; Jules C Hancox
Journal:  J Mol Cell Cardiol       Date:  2019-07-27       Impact factor: 5.000

Review 8.  Rett Syndrome: Crossing the Threshold to Clinical Translation.

Authors:  David M Katz; Adrian Bird; Monica Coenraads; Steven J Gray; Debashish U Menon; Benjamin D Philpot; Daniel C Tarquinio
Journal:  Trends Neurosci       Date:  2016-02       Impact factor: 13.837

Review 9.  Targeted pharmacological treatment of autism spectrum disorders: fragile X and Rett syndromes.

Authors:  Hansen Wang; Sandipan Pati; Lucas Pozzo-Miller; Laurie C Doering
Journal:  Front Cell Neurosci       Date:  2015-02-26       Impact factor: 5.505

10.  Exploring the Molecular Interactions of 7,8-Dihydroxyflavone and Its Derivatives with TrkB and VEGFR2 Proteins.

Authors:  Nitin Chitranshi; Vivek Gupta; Sanjay Kumar; Stuart L Graham
Journal:  Int J Mol Sci       Date:  2015-09-03       Impact factor: 5.923

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