Journal of child neurology · 2012
Lissencephaly and band heterotopia: LIS1, TUBA1A, and DCX mutations in Hungary.
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Mokánszki Attila, Körhegyi Ivett, Szabó Nóra, Bereg Edit, Gergev Gyurgyinka, Balogh Erzsébet, Bessenyei Beáta, Sümegi Andrea, Morris-Rosendahl Deborah J, Sztriha László, Oláh Eva
Аннотация
The spectrum of lissencephaly ranges from absent (agyria) or decreased (pachygyria) convolutions to less severe malformation known as subcortical band heterotopia. Mutations involving LIS1 and TUBA1A result in the classic form of lissencephaly, whereas mutations of the DCX gene cause lissencephaly in males and subcortical band heterotopia in females. This report describes the clinical manifestations and imaging and genetic findings in 2 boys with lissencephaly and a girl with subcortical band heterotopia. An ovel mutation (c.83_84delAT, p.Tyr28Phefs*31) was identified in LIS1 in 1 of the boys with lissencephaly and another novel mutation (c.200delG, p.Ile68Leufs*87) was found in DCX in the girl with subcortical band heterotopia. The mutations appeared in the first half of the genes and are predicted to result in truncated proteins. A mutation was found in the TUBA1A gene (c.1205G>A, p.Arg402His) in the other boy. This mutation affects the folding of tubulin heterodimers, changing the interactions with proteins that bind microtubules.
Предметные рубрики MeSH
1-Alkyl-2-acetylglycerophosphocholine EsteraseBrainChildChild, PreschoolClassical Lissencephalies and Subcortical Band HeterotopiasDNA Mutational AnalysisDoublecortin Domain ProteinsDoublecortin ProteinElectroencephalographyFemaleGenetic Predisposition to DiseaseHumansHungaryMagnetic Resonance ImagingMaleMicrotubule-Associated ProteinsMutationNeuropeptidesPolymorphism, Single NucleotideTubulin