Reanalysis and reclassification of rare genetic variants associated with inherited arrhythmogenic syndromes

Background: Accurate interpretation of rare genetic variants is a challenge for clinical translation. Updates in recommendations for rare variant classification require the reanalysis and reclassification. We aim to perform an exhaustive re-analysis of rare variants associated with inherited arrhyth...

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Detalles Bibliográficos
Autores: Campuzano Larrea, Oscar, Sarquella Brugada, Geòrgia, Fernández-Falgueras, Anna, Coll Vidal, Mònica, Iglesias, Anna, Ferrer Costa, Carles, Cesar, Sergi, Arbelo, Elena, García Álvarez, Ana, Jordà, Paloma, Toro, Rocío, Tirón de Llano, Coloma, Grassi, Simone, Oliva, Antonio, Brugada Terradellas, Josep, Brugada, Ramon
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2020
País:España
Institución:Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)
Repositorio:Recercat. Dipósit de la Recerca de Catalunya
OAI Identifier:oai:recercat.cat:10256/19483
Acceso en línea:http://hdl.handle.net/10256/19483
Access Level:acceso abierto
Palabra clave:Arítmia
Arrhythmia
Cor -- Malalties -- Aspectes genètics
Heart -- Diseases -- Genetic aspects
Cor -- Malalties -- Patogènesi
Heart -- Diseases -- Pathogenesis
Mort sobtada
Sudden death
Descripción
Sumario:Background: Accurate interpretation of rare genetic variants is a challenge for clinical translation. Updates in recommendations for rare variant classification require the reanalysis and reclassification. We aim to perform an exhaustive re-analysis of rare variants associated with inherited arrhythmogenic syndromes, which were classified ten years ago, to determine whether their classification aligns with current standards and research findings. Methods: In 2010, the rare variants identified through genetic analysis were classified following recommendations available at that time. Nowadays, the same variants have been reclassified following current American College of Medical Genetics and Genomics recommendations. Findings: Our cohort included 104 cases diagnosed with inherited arrhythmogenic syndromes and 17 post-mortem cases in which inherited arrhythmogenic syndromes was cause of death. 71.87% of variants change their classification. While 65.62% of variants were classified as likely pathogenic in 2010, after reanalysis, only 17.96% remain as likely pathogenic. In 2010, 18.75% of variants were classified as uncertain role but nowadays 60.15% of variants are classified of unknown significance. Interpretation: Reclassification occurred in more than 70% of rare variants associated with inherited arrhythmogenic syndromes. Our results support the periodical reclassification and personalized clinical translation of rare variants to improve diagnosis and adjust treatment