Literature DB >> 26830113

Rett Syndrome: Crossing the Threshold to Clinical Translation.

David M Katz1, Adrian Bird2, Monica Coenraads3, Steven J Gray4, Debashish U Menon5, Benjamin D Philpot6, Daniel C Tarquinio7.   

Abstract

Lying at the intersection between neurobiology and epigenetics, Rett syndrome (RTT) has garnered intense interest in recent years, not only from a broad range of academic scientists, but also from the pharmaceutical and biotechnology industries. In addition to the critical need for treatments for this devastating disorder, optimism for developing RTT treatments derives from a unique convergence of factors, including a known monogenic cause, reversibility of symptoms in preclinical models, a strong clinical research infrastructure highlighted by an NIH-funded natural history study and well-established clinics with significant patient populations. Here, we review recent advances in understanding the biology of RTT, particularly promising preclinical findings, lessons from past clinical trials, and critical elements of trial design for rare disorders.
Copyright © 2016. Published by Elsevier Ltd.

Entities:  

Keywords:  MECP2; clinical trials; epigenetics; gene therapy; neurodevelopmental disorders; preclinical models

Mesh:

Year:  2016        PMID: 26830113      PMCID: PMC4924590          DOI: 10.1016/j.tins.2015.12.008

Source DB:  PubMed          Journal:  Trends Neurosci        ISSN: 0166-2236            Impact factor:   13.837


  124 in total

1.  MeCP2 binds to non-CG methylated DNA as neurons mature, influencing transcription and the timing of onset for Rett syndrome.

Authors:  Lin Chen; Kaifu Chen; Laura A Lavery; Steven Andrew Baker; Chad A Shaw; Wei Li; Huda Y Zoghbi
Journal:  Proc Natl Acad Sci U S A       Date:  2015-04-13       Impact factor: 11.205

2.  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

Review 3.  Astrocytes conspire with neurons during progression of neurological disease.

Authors:  James C McGann; Daniel T Lioy; Gail Mandel
Journal:  Curr Opin Neurobiol       Date:  2012-04-03       Impact factor: 6.627

4.  Partial rescue of Rett syndrome by ω-3 polyunsaturated fatty acids (PUFAs) oil.

Authors:  Claudio De Felice; Cinzia Signorini; Thierry Durand; Lucia Ciccoli; Silvia Leoncini; Maurizio D'Esposito; Stefania Filosa; Camille Oger; Alexandre Guy; Valérie Bultel-Poncé; Jean-Marie Galano; Alessandra Pecorelli; Laura De Felice; Giuseppe Valacchi; Joussef Hayek
Journal:  Genes Nutr       Date:  2012-03-08       Impact factor: 5.523

5.  Neurobiology of Rett syndrome.

Authors:  M V Johnston; C Hohmann; M E Blue
Journal:  Neuropediatrics       Date:  1995-04       Impact factor: 1.947

Review 6.  The neurobiology of Rett syndrome.

Authors:  Schahram Akbarian
Journal:  Neuroscientist       Date:  2003-02       Impact factor: 7.519

7.  Loss of silent-chromatin looping and impaired imprinting of DLX5 in Rett syndrome.

Authors:  Shin-ichi Horike; Shutao Cai; Masaru Miyano; Jan-Fang Cheng; Terumi Kohwi-Shigematsu
Journal:  Nat Genet       Date:  2004-12-19       Impact factor: 38.330

8.  Systemic delivery of MeCP2 rescues behavioral and cellular deficits in female mouse models of Rett syndrome.

Authors:  Saurabh K Garg; Daniel T Lioy; Hélène Cheval; James C McGann; John M Bissonnette; Matthew J Murtha; Kevin D Foust; Brian K Kaspar; Adrian Bird; Gail Mandel
Journal:  J Neurosci       Date:  2013-08-21       Impact factor: 6.167

9.  Adult neural function requires MeCP2.

Authors:  Christopher M McGraw; Rodney C Samaco; Huda Y Zoghbi
Journal:  Science       Date:  2011-06-02       Impact factor: 47.728

Review 10.  Preclinical research in Rett syndrome: setting the foundation for translational success.

Authors:  David M Katz; Joanne E Berger-Sweeney; James H Eubanks; Monica J Justice; Jeffrey L Neul; Lucas Pozzo-Miller; Mary E Blue; Diana Christian; Jacqueline N Crawley; Maurizio Giustetto; Jacky Guy; C James Howell; Miriam Kron; Sacha B Nelson; Rodney C Samaco; Laura R Schaevitz; Coryse St Hillaire-Clarke; Juan L Young; Huda Y Zoghbi; Laura A Mamounas
Journal:  Dis Model Mech       Date:  2012-11       Impact factor: 5.758

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  60 in total

1.  The course of awake breathing disturbances across the lifespan in Rett syndrome.

Authors:  Daniel C Tarquinio; Wei Hou; Jeffrey L Neul; Gamze Kilic Berkmen; Jana Drummond; Elizabeth Aronoff; Jennifer Harris; Jane B Lane; Walter E Kaufmann; Kathleen J Motil; Daniel G Glaze; Steven A Skinner; Alan K Percy
Journal:  Brain Dev       Date:  2018-04-12       Impact factor: 1.961

2.  Developmental timing and critical windows for the treatment of psychiatric disorders.

Authors:  Oscar Marín
Journal:  Nat Med       Date:  2016-10-26       Impact factor: 53.440

3.  Modeling Rett Syndrome Using TALEN-Edited MECP2 Mutant Cynomolgus Monkeys.

Authors:  Yongchang Chen; Juehua Yu; Yuyu Niu; Dongdong Qin; Hailiang Liu; Gang Li; Yingzhou Hu; Jiaojian Wang; Yi Lu; Yu Kang; Yong Jiang; Kunhua Wu; Siguang Li; Jingkuan Wei; Jing He; Junbang Wang; Xiaojing Liu; Yuping Luo; Chenyang Si; Raoxian Bai; Kunshan Zhang; Jie Liu; Shaoyong Huang; Zhenzhen Chen; Shuang Wang; Xiaoying Chen; Xinhua Bao; Qingping Zhang; Fuxing Li; Rui Geng; Aibin Liang; Dinggang Shen; Tianzi Jiang; Xintian Hu; Yuanye Ma; Weizhi Ji; Yi Eve Sun
Journal:  Cell       Date:  2017-05-18       Impact factor: 41.582

4.  Autonomic dysfunction and sudden death in patients with Rett syndrome: a systematic review

Authors:  Jatinder Singh; Evamaria Lanzarini; Paramala Santosh
Journal:  J Psychiatry Neurosci       Date:  2020-05-01       Impact factor: 6.186

5.  A Psychometric Evaluation of the Motor-Behavioral Assessment Scale for Use as an Outcome Measure in Rett Syndrome Clinical Trials.

Authors:  Melissa Raspa; Carla M Bann; Angela Gwaltney; Timothy A Benke; Cary Fu; Daniel G Glaze; Richard Haas; Peter Heydemann; Mary Jones; Walter E Kaufmann; David Lieberman; Eric Marsh; Sarika Peters; Robin Ryther; Shannon Standridge; Steven A Skinner; Alan K Percy; Jeffrey L Neul
Journal:  Am J Intellect Dev Disabil       Date:  2020-11-01

6.  The Pathophysiology of Rett Syndrome With a Focus on Breathing Dysfunctions.

Authors:  Jan-Marino Ramirez; Marlusa Karlen-Amarante; Jia-Der Ju Wang; Nicholas E Bush; Michael S Carroll; Debra E Weese-Mayer; Alyssa Huff
Journal:  Physiology (Bethesda)       Date:  2020-11-01

7.  Neurobiology: Domains to the rescue for Rett syndrome.

Authors:  Anne E West
Journal:  Nature       Date:  2017-10-11       Impact factor: 49.962

Review 8.  Intellectual and developmental disabilities research centers: Fifty years of scientific accomplishments.

Authors:  Steven U Walkley; Leonard Abbeduto; Mark L Batshaw; Anita Bhattacharyya; Susan Y Bookheimer; Bradley T Christian; John N Constantino; Jean de Vellis; Daniel A Doherty; David L Nelson; Joseph Piven; Annapurna Poduri; Scott L Pomeroy; Rodney C Samaco; Huda Y Zoghbi; Michael J Guralnick
Journal:  Ann Neurol       Date:  2019-07-27       Impact factor: 10.422

9.  A mixed modality approach towards Xi reactivation for Rett syndrome and other X-linked disorders.

Authors:  Lieselot L G Carrette; Chen-Yu Wang; Chunyao Wei; William Press; Weiyuan Ma; Raymond J Kelleher; Jeannie T Lee
Journal:  Proc Natl Acad Sci U S A       Date:  2017-12-27       Impact factor: 11.205

10.  Tsix-Mecp2 female mouse model for Rett syndrome reveals that low-level MECP2 expression extends life and improves neuromotor function.

Authors:  Lieselot L G Carrette; Roy Blum; Weiyuan Ma; Raymond J Kelleher; Jeannie T Lee
Journal:  Proc Natl Acad Sci U S A       Date:  2018-07-23       Impact factor: 11.205

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