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Novel variant in HDAC8 gene resulting in the severe Cornelia de Lange phenotype.
Jezela-Stanek, Aleksandra; Murcia, Pienkowski Victor; Jurkiewicz, Dorota; Iwanicka-Pronicka, Katarzyna; Jedrzejowska, Maria; Krajewska-Walasek, Malgorzata; Ploski, Rafal.
Afiliación
  • Jezela-Stanek A; Department of Genetics and Clinical Immunology, National Institute of Tuberculosis and Lung Diseases.
  • Murcia PV; Department of Medical Genetics, Warsaw Medical University.
  • Jurkiewicz D; Postgraduate School of Molecular Medicine.
  • Iwanicka-Pronicka K; Department of Medical Genetics, The Children's Memorial Health Institute.
  • Jedrzejowska M; Department of Medical Genetics, The Children's Memorial Health Institute.
  • Krajewska-Walasek M; Department of Audiology and Phoniatry, The Children's Memorial Health Institute.
  • Ploski R; Department of Medical Genetics, The Children's Memorial Health Institute.
Clin Dysmorphol ; 28(3): 126-130, 2019 Jul.
Article en En | MEDLINE | ID: mdl-30921088
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.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Represoras / Síndrome de Cornelia de Lange / Histona Desacetilasas Tipo de estudio: Prognostic_studies Límite: Adolescent / Child / Humans / Male Idioma: En Revista: Clin Dysmorphol Asunto de la revista: TERATOLOGIA Año: 2019 Tipo del documento: Article Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Represoras / Síndrome de Cornelia de Lange / Histona Desacetilasas Tipo de estudio: Prognostic_studies Límite: Adolescent / Child / Humans / Male Idioma: En Revista: Clin Dysmorphol Asunto de la revista: TERATOLOGIA Año: 2019 Tipo del documento: Article Pais de publicación: Reino Unido