Evidence map›Paper›PMID 39503779›Full record

ArticleHuman genetics2024

Interpreting the actionable clinical role of rare variants associated with short QT syndrome.

Estefanía Martínez-Barrios, Andrea Greco, José Cruzalegui, Sergi Cesar, Nuria Díez-Escuté, Patricia Cerralbo, Fredy Chipa, Irene Zschaeck, Leonel Slanovic, Alipio Mangas and 4 more

Abstract read
In one paragraph

Article in Human genetics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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2 · The registry

The trial behind it

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

14 authors.

Estefanía Martínez-BarriosArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Andrea GrecoArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
José CruzaleguiArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Sergi CesarArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Nuria Díez-EscutéArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Patricia CerralboArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Fredy ChipaArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Irene ZschaeckArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Leonel SlanovicArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain.
Alipio MangasMedicine Department, School of Medicine, University of Cádiz, Cádiz, 11002, Spain.
Rocío ToroMedicine Department, School of Medicine, University of Cádiz, Cádiz, 11002, Spain.
Josep BrugadaArrhythmias Unit, Hospital Clinic, University of Barcelona, Barcelona, 08036, Spain.
Georgia Sarquella-BrugadaArrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Esplugues de Llobregat, 08950, Spain. georgia@brugada.org.ORCID http://orcid.org/0000-0002-6857-8904
Oscar CampuzanoMedical Science Department, School of Medicine, Universitat de Girona, C/ Emili Grahit 77, Girona, Catalonia, 17003, Spain. oscar@brugada.org.ORCID http://orcid.org/0000-0001-5298-5276

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Genetic testing is recommended in the diagnosis of short QT syndrome. This rare inherited lethal entity is characterized by structural normal hearts with short QT intervals in the electrocardiogram. Few families diagnosed with this arrhythmogenic disease have been reported worldwide so far, impeding a comprehensive understanding of this syndrome. Unraveling the origin of the disease helps to the early identification of genetic carriers at risk. However, only rare variants with a definite deleterious role should be actionable in clinical practice. Our aim was to perform a comprehensive update and reinterpretation, according to the American College of Medical Genetics and Genomics recommendations of all rare variants currently associated with short QT syndrome. We identified 34 rare variants. Reanalysis showed that only nine variants played a deleterious role associated with a definite short QT syndrome phenotype. These variants were located in the four main genes: KCNQ1, KCNH2, KCNJ2 or SLC4A3. Additional rare variants located in other genes were associated with other conditions with phenotypic shortened QT intervals, but not definite diagnosis of short QT syndrome. Periodically updating of rare variants, especially those previously classified as unknown, helps to clarify the role of rare variants and translate genetic data into clinical practice.

Indexed as

Arrhythmias, CardiacKCNQ1 Potassium ChannelElectrocardiographyERG1 Potassium ChannelGenetic Predisposition to DiseaseGenetic TestingGenetic VariationHumansMutationPhenotypePotassium Channels, Inwardly RectifyingERG1 Potassium ChannelKCNH2 protein, humanKCNJ2 protein, humanKCNQ1 Potassium ChannelKCNQ1 protein, humanPotassium Channels, Inwardly Rectifying

Identifiers

PMID39503779
PMCPMC11576798

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.