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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.
- 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.
- Subjects :
- Adolescent
Child
Child, Preschool
DNA Mutational Analysis
Female
Genetic Association Studies
Genotype
Humans
Models, Molecular
Rett Syndrome physiopathology
Severity of Illness Index
Computational Biology
Genetic Predisposition to Disease genetics
Methyl-CpG-Binding Protein 2 genetics
Mutation, Missense genetics
Rett Syndrome genetics
Subjects
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