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Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants

Short QT syndrome, one of the most lethal entities associated with sudden cardiac death, is a rare genetic disease characterized by short QT intervals detected by electrocardiogram. Several genetic variants are causally linked to the disease, but there has yet to be a comprehensive analysis of varia...

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Autores principales: Campuzano, Oscar, Fernandez-Falgueras, Anna, Lemus, Ximena, Sarquella-Brugada, Georgia, Cesar, Sergi, Coll, Monica, Mates, Jesus, Arbelo, Elena, Jordà, Paloma, Perez-Serra, Alexandra, del Olmo, Bernat, Ferrer-Costa, Carles, Iglesias, Anna, Fiol, Victoria, Puigmulé, Marta, Lopez, Laura, Pico, Ferran, Brugada, Josep, Brugada, Ramon
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6678338/
https://www.ncbi.nlm.nih.gov/pubmed/31315195
http://dx.doi.org/10.3390/jcm8071035
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author Campuzano, Oscar
Fernandez-Falgueras, Anna
Lemus, Ximena
Sarquella-Brugada, Georgia
Cesar, Sergi
Coll, Monica
Mates, Jesus
Arbelo, Elena
Jordà, Paloma
Perez-Serra, Alexandra
del Olmo, Bernat
Ferrer-Costa, Carles
Iglesias, Anna
Fiol, Victoria
Puigmulé, Marta
Lopez, Laura
Pico, Ferran
Brugada, Josep
Brugada, Ramon
author_facet Campuzano, Oscar
Fernandez-Falgueras, Anna
Lemus, Ximena
Sarquella-Brugada, Georgia
Cesar, Sergi
Coll, Monica
Mates, Jesus
Arbelo, Elena
Jordà, Paloma
Perez-Serra, Alexandra
del Olmo, Bernat
Ferrer-Costa, Carles
Iglesias, Anna
Fiol, Victoria
Puigmulé, Marta
Lopez, Laura
Pico, Ferran
Brugada, Josep
Brugada, Ramon
author_sort Campuzano, Oscar
collection PubMed
description Short QT syndrome, one of the most lethal entities associated with sudden cardiac death, is a rare genetic disease characterized by short QT intervals detected by electrocardiogram. Several genetic variants are causally linked to the disease, but there has yet to be a comprehensive analysis of variants among patients with short QT syndrome. To fill this gap, we performed an exhaustive study of variants currently catalogued as deleterious in short QT syndrome according to the American College of Medical Genetics and Genomics and the Association for Molecular Pathology. Analysis of the 32 variants described in the literature determined that only nine (28.12%) have a conclusive pathogenic role. All definitively pathogenic variants are located in KCNQ1, KCNH2, or KCNJ2; three genes encoding potassium channels. Other variants located in genes encoding calcium or sodium channels are associated with electrical alterations concomitant with shortened QT intervals but do not guarantee a diagnosis of short QT syndrome. We recommend caution regarding previously reported variants classified as pathogenic. An exhaustive re-analysis is necessary to clarify the role of each variant before routinely translating genetic findings to the clinical setting.
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spelling pubmed-66783382019-08-19 Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants Campuzano, Oscar Fernandez-Falgueras, Anna Lemus, Ximena Sarquella-Brugada, Georgia Cesar, Sergi Coll, Monica Mates, Jesus Arbelo, Elena Jordà, Paloma Perez-Serra, Alexandra del Olmo, Bernat Ferrer-Costa, Carles Iglesias, Anna Fiol, Victoria Puigmulé, Marta Lopez, Laura Pico, Ferran Brugada, Josep Brugada, Ramon J Clin Med Article Short QT syndrome, one of the most lethal entities associated with sudden cardiac death, is a rare genetic disease characterized by short QT intervals detected by electrocardiogram. Several genetic variants are causally linked to the disease, but there has yet to be a comprehensive analysis of variants among patients with short QT syndrome. To fill this gap, we performed an exhaustive study of variants currently catalogued as deleterious in short QT syndrome according to the American College of Medical Genetics and Genomics and the Association for Molecular Pathology. Analysis of the 32 variants described in the literature determined that only nine (28.12%) have a conclusive pathogenic role. All definitively pathogenic variants are located in KCNQ1, KCNH2, or KCNJ2; three genes encoding potassium channels. Other variants located in genes encoding calcium or sodium channels are associated with electrical alterations concomitant with shortened QT intervals but do not guarantee a diagnosis of short QT syndrome. We recommend caution regarding previously reported variants classified as pathogenic. An exhaustive re-analysis is necessary to clarify the role of each variant before routinely translating genetic findings to the clinical setting. MDPI 2019-07-16 /pmc/articles/PMC6678338/ /pubmed/31315195 http://dx.doi.org/10.3390/jcm8071035 Text en © 2019 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Campuzano, Oscar
Fernandez-Falgueras, Anna
Lemus, Ximena
Sarquella-Brugada, Georgia
Cesar, Sergi
Coll, Monica
Mates, Jesus
Arbelo, Elena
Jordà, Paloma
Perez-Serra, Alexandra
del Olmo, Bernat
Ferrer-Costa, Carles
Iglesias, Anna
Fiol, Victoria
Puigmulé, Marta
Lopez, Laura
Pico, Ferran
Brugada, Josep
Brugada, Ramon
Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title_full Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title_fullStr Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title_full_unstemmed Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title_short Short QT Syndrome: A Comprehensive Genetic Interpretation and Clinical Translation of Rare Variants
title_sort short qt syndrome: a comprehensive genetic interpretation and clinical translation of rare variants
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6678338/
https://www.ncbi.nlm.nih.gov/pubmed/31315195
http://dx.doi.org/10.3390/jcm8071035
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