Literature DB >> 11738864

Mutation analysis of the methyl-CpG-binding protein 2 gene (MECP2) in Rett patients with preserved speech.

Y Yamashita1, I Kondo, T Fukuda, R Morishima, A Kusaga, R Iwanaga, T Matsuishi.   

Abstract

Genomic DNAs from 35 Japanese sporadic patients with Rett syndrome (RTT) were screened for DNA mutations in the entire coding region and exon-intron boundaries of methyl-CpG-binding protein 2 (MECP2). We detected mutations in 30 (85.7%) of 35 patients. Among these 35 RTT patients, five patients (14%) had the preserved speech variant of this disease. Four respective mutations (R133C, R306C, R294X, 2 base pair (bp) deletion) were found in these five patients. Two patients had the same missense mutation, R133C. The patients with the R133C mutation and one with frameshift mutation presented the relatively mild clinical presentation, and the R133C mutation was not found in any other patient without preserved speech. We confirmed that the preserved speech variant is one of the clinical phenotypes of RTT and is also caused by MECP2 mutation. We speculated that the clinical phenotype of patients with the R133C missense mutation might be mild.

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Year:  2001        PMID: 11738864     DOI: 10.1016/s0387-7604(01)00378-3

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  8 in total

Review 1.  Rett syndrome and MeCP2: linking epigenetics and neuronal function.

Authors:  Mona D Shahbazian; Huda Y Zoghbi
Journal:  Am J Hum Genet       Date:  2002-11-19       Impact factor: 11.025

2.  Multiple modes of interaction between the methylated DNA binding protein MeCP2 and chromatin.

Authors:  Tatiana Nikitina; Xi Shi; Rajarshi P Ghosh; Rachel A Horowitz-Scherer; Jeffrey C Hansen; Christopher L Woodcock
Journal:  Mol Cell Biol       Date:  2006-11-13       Impact factor: 4.272

3.  Macrocephalic mental retardation associated with a novel C-terminal MECP2 frameshift deletion.

Authors:  Konrad Oexle; Barbara Thamm-Mücke; Thomas Mayer; Sigrid Tinschert
Journal:  Eur J Pediatr       Date:  2004-11-19       Impact factor: 3.183

4.  Heterogeneity in residual function of MeCP2 carrying missense mutations in the methyl CpG binding domain.

Authors:  S Kudo; Y Nomura; M Segawa; N Fujita; M Nakao; C Schanen; M Tamura
Journal:  J Med Genet       Date:  2003-07       Impact factor: 6.318

5.  Specific mutations in methyl-CpG-binding protein 2 confer different severity in Rett syndrome.

Authors:  J L Neul; P Fang; J Barrish; J Lane; E B Caeg; E O Smith; H Zoghbi; A Percy; D G Glaze
Journal:  Neurology       Date:  2008-03-12       Impact factor: 9.910

6.  Gross motor profile in rett syndrome as determined by video analysis.

Authors:  J A Downs; A Bebbington; P Jacoby; M E Msall; O McIlroy; S Fyfe; N Bahi-Buisson; W E Kaufmann; H Leonard
Journal:  Neuropediatrics       Date:  2009-01-22       Impact factor: 1.947

7.  CDKL5/STK9 is mutated in Rett syndrome variant with infantile spasms.

Authors:  E Scala; F Ariani; F Mari; R Caselli; C Pescucci; I Longo; I Meloni; D Giachino; M Bruttini; G Hayek; M Zappella; A Renieri
Journal:  J Med Genet       Date:  2005-02       Impact factor: 6.318

8.  Analysis of X-inactivation status in a Rett syndrome natural history study cohort.

Authors:  Xiaolan Fang; Kameryn M Butler; Fatima Abidi; Jennifer Gass; Arthur Beisang; Timothy Feyma; Robin C Ryther; Shannon Standridge; Peter Heydemann; Mary Jones; Richard Haas; David N Lieberman; Eric D Marsh; Tim A Benke; Steve Skinner; Jeffrey L Neul; Alan K Percy; Michael J Friez; Raymond C Caylor
Journal:  Mol Genet Genomic Med       Date:  2022-03-23       Impact factor: 2.473

  8 in total

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