Literature DB >> 28399682

Clinical, Molecular, and Computational Analysis in Patients With a Novel Double Mutation and a New Synonymous Variant in MeCP2: Report of the First Missense Mutation Within the AT-hook1 Cluster in Rett Syndrome.

Marwa Kharrat1,2, Yosra Kamoun3, Fatma Kamoun4, Emna Ellouze4, Marwa Maalej2, Nourhene Fendri-Kriaa1, Leila Ammar-Keskes2, Neila Belghith2, Ali Gargouri3, Chahnez Triki4, Faiza Fakhfakh1.   

Abstract

Rett syndrome is an X-linked neurodevelopmental disorder, primarily caused by MECP2 mutations. In this study, clinical, molecular and bioinformatics analyses were performed in Rett patients to understand the relationship between MECP2 mutation type and the clinical severity. Two double MeCP2 mutations were detected: a novel one (p.G185 V in cis with p.R255X) in P1 and a known one (p.P179 S in cis with p.R255X) in P2. Besides, a novel synonymous mutation (c.807C>T; p.G269G), which could affect mRNA splicing, was identified in P3. The results from clinical severity analysis have shown that P1 was more severely affected than P2 with CSS being 35 and 14, respectively. Therefore, the phenotypic variability in P1 and P2 could be explained by the potential pathogenic effect of the RTT-causing missense mutation p.G185 V in the AT-hook1. In conclusion, clinical, molecular, and in silico investigations in the studied patients have been proven to be substantial for the genotype-phenotype correlation.

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Keywords:  AT-hook1; MECP2; Rett syndrome; chromosome X inactivation; double MECP2 mutation; p.G185 V

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Year:  2017        PMID: 28399682     DOI: 10.1177/0883073817701622

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  1 in total

1.  Detection of Rare Methyl-CpG Binding Protein 2 Gene Missense Mutations in Patients With Schizophrenia.

Authors:  Chia-Hsiang Chen; Min-Chih Cheng; Ailing Huang; Tsung-Ming Hu; Lieh-Yung Ping; Yu-Syuan Chang
Journal:  Front Genet       Date:  2020-05-08       Impact factor: 4.599

  1 in total

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