Literature DB >> 30921088

Novel variant in HDAC8 gene resulting in the severe Cornelia de Lange phenotype.

Aleksandra Jezela-Stanek1, Pienkowski Victor Murcia2,3, Dorota Jurkiewicz4, Katarzyna Iwanicka-Pronicka4,5, Maria Jędrzejowska4,6, Małgorzata Krajewska-Walasek4, Rafał Płoski2.   

Abstract

Cornelia de Lange syndrome (CDLS) is a clinically and genetically heterogeneous developmental disorder characterized by multiple malformations. Primarily, affected individuals have unique and recognizable dysmorphic facial features, cleft palate, distal limb defects, growth retardation, and developmental delay. However, also milder, as well as slightly phenotypically different forms exist. We described herein a patient with CDLS5, an X-linked form, caused by mutations in the HDAC8 gene inherited form the mosaic mother. Analysis of results from whole exome sequencing identified two variants with possible impact on the phenotype. Of them, hemizygous variant (c.938G>A, p.Arg313Gln) inherited from the mosaic mother, was further proved to lead to disease in the proband. Our intention was to delineate this syndrome but also point out the clinical course of the disease, which only in combination with a facial phenotype allow for verification of exome sequencing result.

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Year:  2019        PMID: 30921088     DOI: 10.1097/MCD.0000000000000277

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  2 in total

1.  A Novel Intragenic Duplication in the HDAC8 Gene Underlying a Case of Cornelia de Lange Syndrome.

Authors:  Cristina Lucia-Campos; Irene Valenzuela; Ana Latorre-Pellicer; David Ros-Pardo; Marta Gil-Salvador; María Arnedo; Beatriz Puisac; Neus Castells; Alberto Plaja; Anna Tenes; Ivon Cuscó; Laura Trujillano; Feliciano J Ramos; Eduardo F Tizzano; Paulino Gómez-Puertas; Juan Pié
Journal:  Genes (Basel)       Date:  2022-08-08       Impact factor: 4.141

2.  A novel de novo HDAC8 missense mutation causing Cornelia de Lange syndrome.

Authors:  Catia Mio; Nadia Passon; Federico Fogolari; Claudia Cesario; Antonio Novelli; Carla Pittini; Giuseppe Damante
Journal:  Mol Genet Genomic Med       Date:  2021-08-03       Impact factor: 2.183

  2 in total

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