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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.

Authors :
Kharrat M
Kamoun Y
Kamoun F
Ellouze E
Maalej M
Fendri-Kriaa N
Ammar-Keskes L
Belghith N
Gargouri A
Triki C
Fakhfakh F
Source :
Journal of child neurology [J Child Neurol] 2017 Jul; Vol. 32 (8), pp. 694-703. Date of Electronic Publication: 2017 Apr 11.
Publication Year :
2017

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.

Details

Language :
English
ISSN :
1708-8283
Volume :
32
Issue :
8
Database :
MEDLINE
Journal :
Journal of child neurology
Publication Type :
Academic Journal
Accession number :
28399682
Full Text :
https://doi.org/10.1177/0883073817701622