Literature DB >> 25669997

Dysregulated brain immunity and neurotrophin signaling in Rett syndrome and autism spectrum disorders.

Theoharis C Theoharides1, Marianna Athanassiou2, Smaro Panagiotidou2, Robert Doyle3.   

Abstract

Rett syndrome is a neurodevelopmental disorder, which occurs in about 1:15,000 females and presents with neurologic and communication defects. It is transmitted as an X-linked dominant linked to mutations of the methyl-CpG-binding protein (MeCP2), a gene transcription suppressor, but its definitive pathogenesis is unknown thus hindering development of effective treatments. Almost half of children with Rett syndrome also have behavioral symptoms consistent with those of autism spectrum disorders (ASDs). PubMed was searched (2005-2014) using the terms: allergy, atopy, brain, brain-derived neurotrophic factor (BDNF), corticotropin-releasing hormone (CRH), cytokines, gene mutations, inflammation, mast cells (MCs), microglia, mitochondria, neurotensin (NT), neurotrophins, seizures, stress, and treatment. There are a number of intriguing differences and similarities between Rett syndrome and ASDs. Rett syndrome occurs in females, while ASDs more often in males, and the former has neurologic disabilities unlike ASDs. There is evidence of dysregulated immune system early in life in both conditions. Lack of microglial phagocytosis and decreased levels of BDNF appear to distinguish Rett syndrome from ASDs, in which there is instead microglia activation and/or proliferation and possibly defective BDNF signaling. Moreover, brain mast cell (MC) activation and focal inflammation may be more prominent in ASDs than Rett syndrome. The flavonoid luteolin blocks microglia and MC activation, provides BDNF-like activity, reverses Rett phenotype in mouse models, and has a significant benefit in children with ASDs. Appropriate formulations of luteolin or other natural molecules may be useful in the treatment of Rett syndrome.
Copyright © 2014 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Brain-derived neurotrophic factor; Inflammation; Mast cells; Microglia; Mitochondria; Rett syndrome; Treatment

Mesh:

Substances:

Year:  2014        PMID: 25669997     DOI: 10.1016/j.jneuroim.2014.12.003

Source DB:  PubMed          Journal:  J Neuroimmunol        ISSN: 0165-5728            Impact factor:   3.478


  18 in total

Review 1.  Altered trajectories of neurodevelopment and behavior in mouse models of Rett syndrome.

Authors:  Elizabeth S Smith; Dani R Smith; Charlotte Eyring; Maria Braileanu; Karen S Smith-Connor; Yew Ei Tan; Amanda Y Fowler; Gloria E Hoffman; Michael V Johnston; Sujatha Kannan; Mary E Blue
Journal:  Neurobiol Learn Mem       Date:  2018-11-29       Impact factor: 2.877

Review 2.  Gut-Microbiota-Brain Axis and Its Effect on Neuropsychiatric Disorders With Suspected Immune Dysregulation.

Authors:  Anastasia I Petra; Smaro Panagiotidou; Erifili Hatziagelaki; Julia M Stewart; Pio Conti; Theoharis C Theoharides
Journal:  Clin Ther       Date:  2015-05-01       Impact factor: 3.393

Review 3.  Behavioral phenotypes of genetic mouse models of autism.

Authors:  T M Kazdoba; P T Leach; J N Crawley
Journal:  Genes Brain Behav       Date:  2015-10-22       Impact factor: 3.449

4.  Tagging methyl-CpG-binding domain proteins reveals different spatiotemporal expression and supports distinct functions.

Authors:  Kathleen H Wood; Brian S Johnson; Sarah A Welsh; Jun Y Lee; Yue Cui; Elizabeth Krizman; Edward S Brodkin; Julie A Blendy; Michael B Robinson; Marisa S Bartolomei; Zhaolan Zhou
Journal:  Epigenomics       Date:  2016-04-12       Impact factor: 4.778

Review 5.  Drug and gene delivery across the blood-brain barrier with focused ultrasound.

Authors:  Kelsie F Timbie; Brian P Mead; Richard J Price
Journal:  J Control Release       Date:  2015-09-08       Impact factor: 9.776

Review 6.  Brain in flames - animal models of psychosis: utility and limitations.

Authors:  Daniele Mattei; Regina Schweibold; Susanne A Wolf
Journal:  Neuropsychiatr Dis Treat       Date:  2015-05-27       Impact factor: 2.570

7.  Paradoxical physiological responses to propranolol in a Rett syndrome patient: a case report.

Authors:  P J Santosh; L Bell; K Lievesley; J Singh; F Fiori
Journal:  BMC Pediatr       Date:  2016-11-29       Impact factor: 2.125

8.  Knowledge-Guided Bioinformatics Model for Identifying Autism Spectrum Disorder Diagnostic MicroRNA Biomarkers.

Authors:  Li Shen; Yuxin Lin; Zhandong Sun; Xuye Yuan; Luonan Chen; Bairong Shen
Journal:  Sci Rep       Date:  2016-12-21       Impact factor: 4.379

Review 9.  Atopic diseases and inflammation of the brain in the pathogenesis of autism spectrum disorders.

Authors:  T C Theoharides; I Tsilioni; A B Patel; R Doyle
Journal:  Transl Psychiatry       Date:  2016-06-28       Impact factor: 6.222

10.  Development of the Tailored Rett Intervention and Assessment Longitudinal (TRIAL) database and the Rett Evaluation of Symptoms and Treatments (REST) Questionnaire.

Authors:  Paramala Santosh; Kate Lievesley; Federico Fiori; Jatinder Singh
Journal:  BMJ Open       Date:  2017-06-21       Impact factor: 2.692

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