Literature DB >> 17905670

Oral treatment with desipramine improves breathing and life span in Rett syndrome mouse model.

Sébastien Zanella1, Saida Mebarek, Anne-Marie Lajard, Nathalie Picard, Mathias Dutschmann, Gérard Hilaire.   

Abstract

Rett syndrome is a neurodevelopmental disease due to Mecp2 gene mutations that is associated to complex neurological symptoms, with bioaminergic deficits and life-threatening apneas related to sudden and unexpected death. In male mice, Mecp2-deficiency similarly induces medullary bioaminergic deficits, severe apneas and short life span. Here, we show that long-term oral treatment of Mecp2-deficient male mice with desipramine, an old drug of clinical use known to block norepinephrine uptake and to strengthen its synaptic effects, significantly alleviates their breathing symptoms and prolongs their life span. Although these mouse results identify desipramine as the first oral pharmacological treatment potentially able to alleviate breathing symptoms of Rett syndrome, we recommend further studies of desipramine effects in Mecp2-deficient mice before attempting any clinical trials in Rett patients.

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Year:  2007        PMID: 17905670     DOI: 10.1016/j.resp.2007.08.009

Source DB:  PubMed          Journal:  Respir Physiol Neurobiol        ISSN: 1569-9048            Impact factor:   1.931


  40 in total

1.  A TrkB small molecule partial agonist rescues TrkB phosphorylation deficits and improves respiratory function in a mouse model of Rett syndrome.

Authors:  Danielle A Schmid; Tao Yang; Michael Ogier; Ian Adams; Yatin Mirakhur; Qifang Wang; Stephen M Massa; Frank M Longo; David M Katz
Journal:  J Neurosci       Date:  2012-02-01       Impact factor: 6.167

2.  MeCP2 is critical within HoxB1-derived tissues of mice for normal lifespan.

Authors:  Christopher S Ward; E Melissa Arvide; Teng-Wei Huang; Jong Yoo; Jeffrey L Noebels; Jeffrey L Neul
Journal:  J Neurosci       Date:  2011-07-13       Impact factor: 6.167

Review 3.  Breathing dysfunction in Rett syndrome: understanding epigenetic regulation of the respiratory network.

Authors:  Michael Ogier; David M Katz
Journal:  Respir Physiol Neurobiol       Date:  2008-12-10       Impact factor: 1.931

4.  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

Review 5.  Rett syndrome and MeCP2.

Authors:  Vichithra R B Liyanage; Mojgan Rastegar
Journal:  Neuromolecular Med       Date:  2014-03-11       Impact factor: 3.843

Review 6.  Evaluation of current pharmacological treatment options in the management of Rett syndrome: from the present to future therapeutic alternatives.

Authors:  Christopher A Chapleau; Jane Lane; Lucas Pozzo-Miller; Alan K Percy
Journal:  Curr Clin Pharmacol       Date:  2013-11

Review 7.  Emerging pharmacotherapies for neurodevelopmental disorders.

Authors:  Daniel Z Wetmore; Craig C Garner
Journal:  J Dev Behav Pediatr       Date:  2010-09       Impact factor: 2.225

Review 8.  Breathing abnormalities in animal models of Rett syndrome a female neurogenetic disorder.

Authors:  Chun Jiang; Ningren Cui; Weiwei Zhong; Christopher M Johnson; Yang Wu
Journal:  Respir Physiol Neurobiol       Date:  2016-11-21       Impact factor: 1.931

9.  Defects in brainstem neurons associated with breathing and motor function in the Mecp2R168X/Y mouse model of Rett syndrome.

Authors:  Christopher M Johnson; Weiwei Zhong; Ningren Cui; Yang Wu; Hao Xing; Shuang Zhang; Chun Jiang
Journal:  Am J Physiol Cell Physiol       Date:  2016-09-21       Impact factor: 4.249

Review 10.  Bioaminergic neuromodulation of respiratory rhythm in vitro.

Authors:  Jean-Charles Viemari; Andrew K Tryba
Journal:  Respir Physiol Neurobiol       Date:  2009-08-31       Impact factor: 1.931

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