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De novo MECP2 duplication in two females with random X-inactivation and moderate mental retardation.

Authors :
Grasshoff U
Bonin M
Goehring I
Ekici A
Dufke A
Cremer K
Wagner N
Rossier E
Jauch A
Walter M
Bauer C
Bauer P
Horber K
Beck-Woedl S
Wieczorek D
Source :
European journal of human genetics : EJHG [Eur J Hum Genet] 2011 May; Vol. 19 (5), pp. 507-12. Date of Electronic Publication: 2011 Feb 16.
Publication Year :
2011

Abstract

Xq28 duplications including MECP2 are a well-known cause of severe mental retardation in males with seizures, muscular hypotonia, progressive spasticity, poor speech and recurrent infections that often lead to early death. Female carriers usually show a normal intellectual performance due to skewed X-inactivation (XCI). We report on two female patients with a de novo MECP2 duplication associated with moderate mental retardation. In both patients, the de novo duplication occurred on the paternal allele, and both patients show a random XCI, which can be assumed as the triggering factor for the phenotype. Furthermore, we describe the phenotype that might be restricted to unspecific mild-to -moderate mental retardation with neurological features in early adulthood.

Details

Language :
English
ISSN :
1476-5438
Volume :
19
Issue :
5
Database :
MEDLINE
Journal :
European journal of human genetics : EJHG
Publication Type :
Academic Journal
Accession number :
21326285
Full Text :
https://doi.org/10.1038/ejhg.2010.226