| Literature DB >> 28399682 |
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.Entities:
Keywords: AT-hook1; MECP2; Rett syndrome; chromosome X inactivation; double MECP2 mutation; p.G185 V
Mesh:
Substances:
Year: 2017 PMID: 28399682 DOI: 10.1177/0883073817701622
Source DB: PubMed Journal: J Child Neurol ISSN: 0883-0738 Impact factor: 1.987