| Literature DB >> 32627382 |
Maria Teresa Carminho-Rodrigues1, Dora Steel2, Sergio B Sousa3, Gregor Brandt4, Michel Guipponi1, Sacha Laurent1, Siv Fokstuen1, Aurea Moren5, André Zacharia5, Elisabeth Dirren5, Renata Oliveira3,6, Manju A Kurian2, Pierre R Burkhard5, Julien F Bally5.
Abstract
YY1 mutations cause Gabriele-de Vries syndrome, a recently described condition involving cognitive impairment, facial dysmorphism and intrauterine growth restriction. Movement disorders were reported in 5/10 cases of the original series, but no detailed description was provided. Here we present a 21-year-old woman with a mild intellectual deficit, facial dysmorphism and a complex movement disorder including an action tremor, cerebellar ataxia, dystonia, and partial ocular apraxia as the presenting and most striking feature. Whole-exome sequencing revealed a novel heterozygous de novo mutation in YY1 [NM: 003403.4 (YY1): c.907 T > C; p.(Cys303Arg)], classified as pathogenic according to the ACMG guidelines.Entities:
Keywords: YY1 gene; action-tremor; ataxia; autosomal dominant; dystonia
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Year: 2020 PMID: 32627382 DOI: 10.1002/ajmg.a.61731
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802