Literature DB >> 22750798

Detection of somatic and germline mosaicism for the LAMP2 gene mutation c.808dupG in a Chinese family with Danon disease.

Xiao-Ling Chen1, Yan Zhao, Hai-Ping Ke, Wen-Ting Liu, Zhen-Fang Du, Xian-Ning Zhang.   

Abstract

Danon disease is a rare X-linked lysosomal storage disease characterized by hypertrophic cardiomyopathy, myopathy and mental retardation, and is due to a primary defect in lysosome-associated membrane protein-2 (LAMP 2). More than 26 mutations in the LAMP2 gene have been described, including a small number of de novo mutations, some of which are suspected to be caused by germline mosaicism. Here, we describe the first molecularly documented evidence of somatic mosaicism for a LAMP2 mutation, identified in the asymptomatic mother of a boy with Danon disease caused by the frameshift mutation c.808dupG (p.A270Gfx3) within exon 6. In addition, in order to gain insight into the possible explanation for the mother's lack of phenotype, the level of somatic mosaicism and the X-chromosome inactivation pattern were investigated. This study provides new insight into the causes of phenotypic variability in female mutation-carriers and underlines the importance of parental molecular testing for accurate genetic counseling for Danon disease.
Copyright © 2012 Elsevier B.V. All rights reserved.

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Year:  2012        PMID: 22750798     DOI: 10.1016/j.gene.2012.06.064

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  4 in total

1.  Mosaic tissue distribution of the tandem duplication of LAMP2 exons 4 and 5 demonstrates the limits of Danon disease cellular and molecular diagnostics.

Authors:  Filip Majer; Ondrej Pelak; Tomas Kalina; Hana Vlaskova; Lenka Dvorakova; Tomas Honzik; Tomas Palecek; Petr Kuchynka; Martin Masek; Jiri Zeman; Milan Elleder; Jakub Sikora
Journal:  J Inherit Metab Dis       Date:  2013-05-29       Impact factor: 4.982

Review 2.  Dosage Compensation in Females with X-Linked Metabolic Disorders.

Authors:  Patrycja Juchniewicz; Ewa Piotrowska; Anna Kloska; Magdalena Podlacha; Jagoda Mantej; Grzegorz Węgrzyn; Stefan Tukaj; Joanna Jakóbkiewicz-Banecka
Journal:  Int J Mol Sci       Date:  2021-04-26       Impact factor: 5.923

3.  A family with Danon disease caused by a splice site mutation in LAMP2 that generates a truncated protein.

Authors:  Nianwei Zhou; Jie Cui; Weipeng Zhao; Yingying Jiang; Wenqing Zhu; Lu Tang; Xuejie Li; Minmin Sun; Cuizhen Pan; Xianhong Shu
Journal:  Mol Genet Genomic Med       Date:  2019-02-03       Impact factor: 2.183

4.  Danon disease: Two patients with atrial fibrillation in a single family and review of the literature.

Authors:  Shaohua Guo; Linghuan Zhou; Renping Wang; Zhixin Lv; Hongzun Xu; Baoli Han; Panagiotis Korantzopoulos; Fuli Hu; Tong Liu
Journal:  Exp Ther Med       Date:  2019-07-17       Impact factor: 2.447

  4 in total

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