Personalized Interpretation and Clinical Translation of Genetic Variants Associated With Cardiomyopathies


Por: Campuzano O, Fernandez-Falgueras A, Sarquella-Brugada G, César-Díaz S, Arbelo E, García-Álvarez A, Jordà P, Coll M, Fiol JV, Iglesias A, Perez-Serra A, Mates J, Del Olmo B, Ferrer C, Alcalde M, Puigmulé M, Mademont-Soler I, Pico F, Lopez L, Tiron C, Brugada-Terradellas J and Brugada R

Publicada: 15 may 2019 Ahead of Print: 15 may 2019
Resumen:
Cardiomyopathies are a heterogeneous group of inherited cardiac diseases characterized by progressive myocardium abnormalities associated with mechanical and/or electrical dysfunction. Massive genetic sequencing technologies allow a comprehensive genetic analysis to unravel the cause of disease. However, most identified genetic variants remain of unknown clinical significance due to incomplete penetrance and variable expressivity. Therefore, genetic interpretation of variants and translation into clinical practice remain a current challenge. We performed retrospective comprehensive clinical assessment and genetic analysis in six families, four diagnosed with arrhythmogenic cardiomyopathy, and two diagnosed with hypertrophic cardiomyopathy (HCM). Genetic testing identified three rare variants (two non-sense and one small indel inducing a frameshift), each present in two families. Although each variant is currently classified as pathogenic and the cause of the diagnosed cardiomyopathy, the onset and/or clinical course differed in each patient. New genetic technology allows comprehensive yet cost-effective genetic analysis, although genetic interpretation, and clinical translation of identified variants should be carefully done in each family in a personalized manner.

Filiaciones:
Campuzano O:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

 Department of Medical Science, School of Medicine, University of Girona, Girona, Spain

 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

 Department of Biochemistry and Molecular Genetics, Hospital Clinic, Barcelona, Spain

Fernandez-Falgueras A:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Sarquella-Brugada G:
 Arrhythmias Unit, Hospital Sant Joan de Déu, University of Barcelona, Barcelona, Spain

 Department of Medical Science, School of Medicine, University of Girona, Girona, Spain

César-Díaz S:
 Arrhythmias Unit, Hospital Sant Joan de Déu, University of Barcelona, Barcelona, Spain

Arbelo E:
 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

 Arrhythmias Unit, Hospital Clinic, University of Barcelona, Barcelona, Spain

García-Álvarez A:
 Arrhythmias Unit, Hospital Clinic, University of Barcelona, Barcelona, Spain

Jordà P:
 Arrhythmias Unit, Hospital Clinic, University of Barcelona, Barcelona, Spain

Coll M:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Fiol JV:
 Arrhythmias Unit, Hospital Sant Joan de Déu, University of Barcelona, Barcelona, Spain

Iglesias A:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

Perez-Serra A:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

Mates J:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Del Olmo B:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Ferrer C:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Alcalde M:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

Puigmulé M:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Mademont-Soler I:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Pico F:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Lopez L:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

Tiron C:
 Cardiology Service, Hospital Josep Trueta, University of Girona, Girona, Spain

Brugada-Terradellas J:
 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

 Arrhythmias Unit, Hospital Sant Joan de Déu, University of Barcelona, Barcelona, Spain

 Arrhythmias Unit, Hospital Clinic, University of Barcelona, Barcelona, Spain

Brugada R:
 Cardiovascular Genetics Center, Biomedical Research Institute of Girona, University of Girona, Girona, Spain

 Department of Medical Science, School of Medicine, University of Girona, Girona, Spain

 Centro Investigación Biomédica Red Enfermedades Cardiovasculares, Madrid, Spain

 Cardiology Service, Hospital Josep Trueta, University of Girona, Girona, Spain
ISSN: 16648021





Frontiers in Genetics
Editorial
FRONTIERS MEDIA SA, AVENUE DU TRIBUNAL FEDERAL 34, LAUSANNE CH-1015, SWITZERLAND, Suiza
Tipo de documento: Article
Volumen: 10 Número:
Páginas: 450-450
WOS Id: 000467992100001
ID de PubMed: 31156706
imagen Green Submitted, gold

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