Literature DB >> 19002580

Brain lipid analysis in mice with Rett syndrome.

Thomas N Seyfried1, Karie A Heinecke, John G Mantis, Christine A Denny.   

Abstract

Rett syndrome (RS) is an X-linked neurodevelopmental disorder mostly involving mutations in the gene for methyl-CpG-binding protein 2 (MECP2). Ganglioside abnormalities were previously found in cerebrum and cerebellum in RS patients. We evaluated total lipid distribution in cerebrum/brainstem, hippocampus, and cerebellum in male mice carrying either the Mecp2 (tm1.1Bird) knockout mutation or the Mecp2 (308/y) deletion mutation. The concentration of the neuronal enriched ganglioside GD1a was significantly lower in the cerebrum/brainstem of Mecp2 (tm1.1Bird) mice than in that of age matched controls, but was not reduced in the Mecp2 (308/y) mice. No other differences in brain lipid content, including myelin-enriched cerebrosides, were detected in mice with either type of Mecp2 mutation. These findings indicate that the poor motor performance previously reported in the RS mutant mice is not associated with major brain lipid abnormalities and that most previous brain lipid abnormalities observed in RS patients were not observed in the Mecp2 (tm1.1Bird) or the Mecp2 (308/y) RS mice.

Entities:  

Mesh:

Substances:

Year:  2008        PMID: 19002580      PMCID: PMC4286142          DOI: 10.1007/s11064-008-9874-7

Source DB:  PubMed          Journal:  Neurochem Res        ISSN: 0364-3190            Impact factor:   3.996


  38 in total

1.  Immunocytochemical localization of GD3 ganglioside to astrocytes in murine cerebellar mutants.

Authors:  S M LeVine; T N Seyfried; R K Yu; J E Goldman
Journal:  Brain Res       Date:  1986-05-28       Impact factor: 3.252

2.  A mouse Mecp2-null mutation causes neurological symptoms that mimic Rett syndrome.

Authors:  J Guy; B Hendrich; M Holmes; J E Martin; A Bird
Journal:  Nat Genet       Date:  2001-03       Impact factor: 38.330

3.  Differential cellular enrichment of gangliosides in the mouse cerebellum: analysis using neurological mutants.

Authors:  T N Seyfried; R K Yu; N Miyazawa
Journal:  J Neurochem       Date:  1982-02       Impact factor: 5.372

Review 4.  Rett syndrome. Current status and new vistas.

Authors:  Alan K Percy
Journal:  Neurol Clin       Date:  2002-11       Impact factor: 3.806

5.  Myelin galactolipid synthesis in different strains of mice.

Authors:  C Sato; R K Yu
Journal:  J Neurochem       Date:  1987-10       Impact factor: 5.372

6.  N-butyldeoxygalactonojirimycin reduces brain ganglioside and GM2 content in neonatal Sandhoff disease mice.

Authors:  Rena C Baek; Julie L Kasperzyk; Frances M Platt; Thomas N Seyfried
Journal:  Neurochem Int       Date:  2007-12-08       Impact factor: 3.921

7.  Cellular localization of gangliosides in the mouse cerebellum: analysis using neurological mutants.

Authors:  T N Seyfried; R K Yu
Journal:  Adv Exp Med Biol       Date:  1984       Impact factor: 2.622

8.  Human brain gangliosides: developmental changes from early fetal stage to advanced age.

Authors:  L Svennerholm; K Boström; P Fredman; J E Månsson; B Rosengren; B M Rynmark
Journal:  Biochim Biophys Acta       Date:  1989-09-25

9.  Cellular distribution of gangliosides in the developing mouse cerebellum: analysis using the staggerer mutant.

Authors:  T N Seyfried; D J Bernard; R K Yu
Journal:  J Neurochem       Date:  1984-10       Impact factor: 5.372

Review 10.  Rett syndrome: clinical and molecular update.

Authors:  Alan K Percy; Jane B Lane
Journal:  Curr Opin Pediatr       Date:  2004-12       Impact factor: 2.856

View more
  5 in total

1.  Suppression of brain cholesterol synthesis in male Mecp2-deficient mice is age dependent and not accompanied by a concurrent change in the rate of fatty acid synthesis.

Authors:  Adam M Lopez; Jen-Chieh Chuang; Kenneth S Posey; Stephen D Turley
Journal:  Brain Res       Date:  2016-10-24       Impact factor: 3.252

2.  Vitamin K Deficiency Induced by Warfarin Is Associated With Cognitive and Behavioral Perturbations, and Alterations in Brain Sphingolipids in Rats.

Authors:  Sahar Tamadon-Nejad; Bouchra Ouliass; Joseph Rochford; Guylaine Ferland
Journal:  Front Aging Neurosci       Date:  2018-07-16       Impact factor: 5.750

3.  Changes in the Cerebrospinal Fluid and Plasma Lipidome in Patients with Rett Syndrome.

Authors:  Martina Zandl-Lang; Thomas Züllig; Martin Trötzmüller; Yvonne Naegelin; Lucia Abela; Bernd Wilken; Sabine Scholl-Buergi; Daniela Karall; Ludwig Kappos; Harald Köfeler; Barbara Plecko
Journal:  Metabolites       Date:  2022-03-25

4.  MeCP2 modulates gene expression pathways in astrocytes.

Authors:  Dag H Yasui; Huichun Xu; Keith W Dunaway; Janine M Lasalle; Lee-Way Jin; Izumi Maezawa
Journal:  Mol Autism       Date:  2013-01-25       Impact factor: 7.509

Review 5.  Transcriptomic and Epigenomic Landscape in Rett Syndrome.

Authors:  Domenico Marano; Salvatore Fioriniello; Maurizio D'Esposito; Floriana Della Ragione
Journal:  Biomolecules       Date:  2021-06-30
  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.