Literature DB >> 20814256

Emerging pharmacotherapies for neurodevelopmental disorders.

Daniel Z Wetmore1, Craig C Garner.   

Abstract

A growing and interdisciplinary translational neuroscience research effort for neurodevelopmental disorders (NDDs) is investigating the mechanisms of dysfunction and testing effective treatment strategies in animal models and, when possible, in the clinic. NDDs with a genetic basis have received particular attention. Transgenic animals that mimic genetic insults responsible for disease in man have provided insight about mechanisms of dysfunction, and, surprisingly, have shown that cognitive deficits can be addressed in adult animals. This review will present recent translational research based on animal models of genetic NDDs, as well as pharmacotherapeutic strategies under development to address deficits of brain function for Down syndrome, fragile X syndrome, Rett syndrome, neurofibromatosis-1, tuberous sclerosis, and autism. Although these disorders vary in underlying causes and clinical presentation, common pathways and mechanisms for dysfunction have been observed. These include abnormal gene dosage, imbalance among neurotransmitter systems, and deficits in the development, maintenance and plasticity of neuronal circuits. NDDs affect multiple brain systems and behaviors that may be amenable to drug therapies that target distinct deficits. A primary goal of translational research is to replace symptomatic and supportive drug therapies with pharmacotherapies based on a principled understanding of the causes of dysfunction. Based on this principle, several recently developed therapeutic strategies offer clear promise for clinical development in man.

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Year:  2010        PMID: 20814256      PMCID: PMC2967570          DOI: 10.1097/DBP.0b013e3181ee3833

Source DB:  PubMed          Journal:  J Dev Behav Pediatr        ISSN: 0196-206X            Impact factor:   2.225


  270 in total

1.  The role of different X-inactivation pattern on the variable clinical phenotype with Rett syndrome.

Authors:  T Ishii; Y Makita; A Ogawa; S Amamiya; M Yamamoto; A Miyamoto; J Oki
Journal:  Brain Dev       Date:  2001-12       Impact factor: 1.961

2.  The NF1 tumor suppressor critically regulates TSC2 and mTOR.

Authors:  Cory M Johannessen; Elizabeth E Reczek; Marianne F James; Hilde Brems; Eric Legius; Karen Cichowski
Journal:  Proc Natl Acad Sci U S A       Date:  2005-06-03       Impact factor: 11.205

3.  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

4.  Trisomy for the Down syndrome 'critical region' is necessary but not sufficient for brain phenotypes of trisomic mice.

Authors:  Lisa E Olson; Randall J Roper; Crystal L Sengstaken; Elizabeth A Peterson; Veronica Aquino; Zygmunt Galdzicki; Richard Siarey; Mikhail Pletnikov; Timothy H Moran; Roger H Reeves
Journal:  Hum Mol Genet       Date:  2007-03-05       Impact factor: 6.150

5.  Neuro-epileptic determinants of autism spectrum disorders in tuberous sclerosis complex.

Authors:  Patrick F Bolton; Rebecca J Park; J Nicholas P Higgins; Paul D Griffiths; Andrew Pickles
Journal:  Brain       Date:  2002-06       Impact factor: 13.501

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

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.  Levetiracetam as adjunctive antiepileptic therapy for patients with tuberous sclerosis complex: a retrospective open-label trial.

Authors:  James J Collins; Cynthia Tudor; Jennifer M Leonard; Gail Chuck; David Neal Franz
Journal:  J Child Neurol       Date:  2006-01       Impact factor: 1.987

9.  A placebo-controlled, fixed-dose study of aripiprazole in children and adolescents with irritability associated with autistic disorder.

Authors:  Ronald N Marcus; Randall Owen; Lisa Kamen; George Manos; Robert D McQuade; William H Carson; Michael G Aman
Journal:  J Am Acad Child Adolesc Psychiatry       Date:  2009-11       Impact factor: 8.829

10.  Ube3a mRNA and protein expression are not decreased in Mecp2R168X mutant mice.

Authors:  Amy Lawson-Yuen; Daniel Liu; Liqun Han; Zhichun I Jiang; Guochuan E Tsai; Alo C Basu; Jonathan Picker; Jiamin Feng; Joseph T Coyle
Journal:  Brain Res       Date:  2007-08-24       Impact factor: 3.252

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  16 in total

Review 1.  Trisomy 21 and early brain development.

Authors:  Tarik F Haydar; Roger H Reeves
Journal:  Trends Neurosci       Date:  2011-12-09       Impact factor: 13.837

2.  Parents' decisions to screen newborns for FMR1 gene expansions in a pilot research project.

Authors:  Debra Skinner; Summer Choudhury; John Sideris; Sonia Guarda; Allen Buansi; Myra Roche; Cynthia Powell; Donald B Bailey
Journal:  Pediatrics       Date:  2011-05-29       Impact factor: 7.124

3.  Transcript catalogs of human chromosome 21 and orthologous chimpanzee and mouse regions.

Authors:  Xiaolu Sturgeon; Katheleen J Gardiner
Journal:  Mamm Genome       Date:  2011-03-13       Impact factor: 2.957

4.  Gene expression changes in the MAPK pathway in both Fragile X and Down syndrome human neural progenitor cells.

Authors:  Erin L McMillan; Allison L Kamps; Samuel S Lake; Clive N Svendsen; Anita Bhattacharyya
Journal:  Am J Stem Cells       Date:  2012-06-03

Review 5.  Treatment of neurodevelopmental disorders in adulthood.

Authors:  Eero Castrén; Ype Elgersma; Lamberto Maffei; Randi Hagerman
Journal:  J Neurosci       Date:  2012-10-10       Impact factor: 6.167

6.  Astrocyte-derived thrombospondins mediate the development of hippocampal presynaptic plasticity in vitro.

Authors:  Devon C Crawford; Xiaoping Jiang; Amanda Taylor; Steven Mennerick
Journal:  J Neurosci       Date:  2012-09-19       Impact factor: 6.167

7.  Homozygosity mapping in 64 Syrian consanguineous families with non-specific intellectual disability reveals 11 novel loci and high heterogeneity.

Authors:  R Abou Jamra; Sigrun Wohlfart; Markus Zweier; Steffen Uebe; Lutz Priebe; Arif Ekici; Susanne Giesebrecht; Ahmad Abboud; Mohammed Ayman Al Khateeb; Mahmoud Fakher; Saber Hamdan; Amina Ismael; Safia Muhammad; Markus M Nöthen; Johannes Schumacher; André Reis
Journal:  Eur J Hum Genet       Date:  2011-06-01       Impact factor: 4.246

8.  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

9.  Autism-associated mutations in ProSAP2/Shank3 impair synaptic transmission and neurexin-neuroligin-mediated transsynaptic signaling.

Authors:  Magali H Arons; Charlotte J Thynne; Andreas M Grabrucker; Dong Li; Michael Schoen; Juliette E Cheyne; Tobias M Boeckers; Johanna M Montgomery; Craig C Garner
Journal:  J Neurosci       Date:  2012-10-24       Impact factor: 6.167

Review 10.  Modeling neurodevelopmental cognitive deficits in tasks with cross-species translational validity.

Authors:  Z A Cope; S B Powell; J W Young
Journal:  Genes Brain Behav       Date:  2016-01       Impact factor: 3.449

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