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A new phenotype of severe dilated cardiomyopathy associated with a mutation in the LAMP2 gene previously known to cause hypertrophic cardiomyopathy in the context of Danon disease.
Gourzi, Polyxeni; Pantou, Malena P; Gkouziouta, Angeliki; Kaklamanis, Loukas; Tsiapras, Dimitrios; Zygouri, Christianna; Constantoulakis, Pantelis; Adamopoulos, Stamatis; Degiannis, Dimitrios.
Afiliação
  • Gourzi P; Molecular Immunopathology & Histocompatibility Unit, Division of Molecular Genetics, Onassis Cardiac Surgery Center, Greece.
  • Pantou MP; Molecular Immunopathology & Histocompatibility Unit, Division of Molecular Genetics, Onassis Cardiac Surgery Center, Greece. Electronic address: pantou@ocsc.gr.
  • Gkouziouta A; Heart Failure, MCS & Transplant Unit, Onassis Cardiac Surgery Center, Greece.
  • Kaklamanis L; Department of Pathology, Onassis Cardiac Surgery Center, Greece.
  • Tsiapras D; Division of Cardiology, Onassis Cardiac Surgery Center, Greece.
  • Zygouri C; Department of Molecular Genetics, BioAnalytica-Genotypes SA, Greece.
  • Constantoulakis P; Department of Molecular Genetics, BioAnalytica-Genotypes SA, Greece.
  • Adamopoulos S; Heart Failure, MCS & Transplant Unit, Onassis Cardiac Surgery Center, Greece.
  • Degiannis D; Molecular Immunopathology & Histocompatibility Unit, Division of Molecular Genetics, Onassis Cardiac Surgery Center, Greece.
Eur J Med Genet ; 62(1): 77-80, 2019 Jan.
Article em En | MEDLINE | ID: mdl-29753918
Danon disease is a rare X-linked cardiac and skeletal muscle disorder with multisystem clinical manifestations. Genetic defects at the lysosome-associated membrane 2 protein (LAMP2) are the cause of the disorder. Due to the rarity of the disease, there is limited progress in understanding the correlation between genotype and phenotype, and explaining the large variability of the clinical features of the disease. In this study, we report two patients, twin sisters, referred to our hospital for end stage heart failure due to dilated cardiomyopathy, requiring heart transplant evaluation. Genetic analysis, using targeted next generation sequencing, showed that the proband carried a LAMP2 missense variant, c.928G > A. The mutation was also detected in her twin sister by sanger sequencing. This variant has already been reported by other investigators and was correlated with the clinical triad of Danon disease i.e. hypertrophic cardiomyopathy, mental retardation and peripheral myopathy. The new phenotype of dilated cardiomyopathy associated with this mutation, confirms the phenotypic heterogeneity of the particular mutation, as well as of Danon disease.
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Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Fenótipo / Cardiomiopatia Dilatada / Mutação de Sentido Incorreto / Doença de Depósito de Glicogênio Tipo IIb / Proteína 2 de Membrana Associada ao Lisossomo Tipo de estudo: Risk_factors_studies Limite: Adult / Female / Humans Idioma: En Revista: Eur J Med Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Grécia

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Fenótipo / Cardiomiopatia Dilatada / Mutação de Sentido Incorreto / Doença de Depósito de Glicogênio Tipo IIb / Proteína 2 de Membrana Associada ao Lisossomo Tipo de estudo: Risk_factors_studies Limite: Adult / Female / Humans Idioma: En Revista: Eur J Med Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Grécia