Literature DB >> 32627382

Complex movement disorder in a patient with heterozygous YY1 mutation (Gabriele-de Vries syndrome).

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.
© 2020 Wiley Periodicals LLC.

Entities:  

Keywords:  YY1 gene; action-tremor; ataxia; autosomal dominant; dystonia

Mesh:

Substances:

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


  6 in total

Review 1.  Genetic Dystonias: Update on Classification and New Genetic Discoveries.

Authors:  Ignacio Juan Keller Sarmiento; Niccolò Emanuele Mencacci
Journal:  Curr Neurol Neurosci Rep       Date:  2021-02-09       Impact factor: 5.081

Review 2.  Emerging and converging molecular mechanisms in dystonia.

Authors:  Paulina Gonzalez-Latapi; Nicolas Marotta; Niccolò E Mencacci
Journal:  J Neural Transm (Vienna)       Date:  2021-01-01       Impact factor: 3.575

Review 3.  The apparent paradox of phenotypic diversity and shared mechanisms across dystonia syndromes.

Authors:  Alessio Di Fonzo; Alberto Albanese; Hyder A Jinnah
Journal:  Curr Opin Neurol       Date:  2022-07-05       Impact factor: 6.283

4.  A de novo YY1 missense variant expanding the Gabriele-de Vries syndrome phenotype and affecting X-chromosome inactivation.

Authors:  Suely Rodrigues Dos Santos; Rafael Mina Piergiorge; Jady Rocha; Bianca Barbosa Abdala; Andressa Pereira Gonçalves; Márcia Mattos Gonçalves Pimentel; Cíntia Barros Santos-Rebouças
Journal:  Metab Brain Dis       Date:  2022-07-13       Impact factor: 3.655

5.  A pathogenic DYT-THAP1 dystonia mutation causes hypomyelination and loss of YY1 binding.

Authors:  Dhananjay Yellajoshyula; Abigail E Rogers; Audrey J Kim; Sumin Kim; Samuel S Pappas; William T Dauer
Journal:  Hum Mol Genet       Date:  2022-03-31       Impact factor: 5.121

6.  Untangling neurodevelopmental disorders in the adulthood: a movement disorder is the clue.

Authors:  Elisabetta Indelicato; Michael Zech; Matthias Amprosi; Sylvia Boesch
Journal:  Orphanet J Rare Dis       Date:  2022-02-16       Impact factor: 4.123

  6 in total

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