Literature DB >> 33815637

The Postmortem Interpretation of Cardiac Genetic Variants of Unknown Significance in Sudden Death in the Young: A Case Report and Review of the Literature.

Saleh Fadel, Alfredo E Walker.   

Abstract

Sudden cardiac death (SCD) in adolescents and young adults is a major traumatic event for families and communities. In these cases, it is not uncommon to have a negative autopsy with structurally and histologically normal heart. Such SCD cases are generally attributed to channelopathies, which include long QT syndrome, short QT syndrome, Brugada syndrome, and catecholaminergic polymorphic ventricular tachycardia. Our understanding of the causes for SCDs has changed significantly with the advancements in molecular and genetic studies, where many mutations are now known to be associated with certain channelopathies. Postmortem analysis provides great value in informing decision-making with regard to screening tests and prophylactic measures that should be taken to prevent sudden death in first degree relatives of the decedent. As this is a rapidly advancing field, our ability to identify genetic mutations has surpassed our ability to interpret them. This led to a unique challenge in genetic testing called variants of unknown significance (VUS). VUSs present a diagnostic dilemma and uncertainty for clinicians and patients with regard to next steps. Caution should be exercised when interpreting VUSs since misinterpretation can result in mismanagement of patients and their families. A case of a young adult man with drowning as his proximate cause of death is presented in circumstances where cardiac genetic testing was indicated and undertaken. Eight VUSs in genes implicated in inheritable cardiac dysfunction were identified and the interpretation of VUSs in this scenario is discussed.
© The Author(s) 2021.

Entities:  

Keywords:  Cardiac genetic mutations; Forensic pathology; Sudden cardiac death; Sudden death; Variant of uncertain significance

Year:  2021        PMID: 33815637      PMCID: PMC7975994          DOI: 10.1177/1925362120984868

Source DB:  PubMed          Journal:  Acad Forensic Pathol        ISSN: 1925-3621


  79 in total

1.  Diagnostic yield in sudden unexplained death and aborted cardiac arrest in the young: the experience of a tertiary referral center in The Netherlands.

Authors:  Christian van der Werf; Nynke Hofman; Hanno L Tan; Pascal F van Dessel; Marielle Alders; Allard C van der Wal; Irene M van Langen; Arthur A M Wilde
Journal:  Heart Rhythm       Date:  2010-05-31       Impact factor: 6.343

Review 2.  Genetic purgatory and the cardiac channelopathies: Exposing the variants of uncertain/unknown significance issue.

Authors:  Michael J Ackerman
Journal:  Heart Rhythm       Date:  2015-07-02       Impact factor: 6.343

Review 3.  Brugada syndrome: report of the second consensus conference: endorsed by the Heart Rhythm Society and the European Heart Rhythm Association.

Authors:  Charles Antzelevitch; Pedro Brugada; Martin Borggrefe; Josep Brugada; Ramon Brugada; Domenico Corrado; Ihor Gussak; Herve LeMarec; Koonlawee Nademanee; Andres Ricardo Perez Riera; Wataru Shimizu; Eric Schulze-Bahr; Hanno Tan; Arthur Wilde
Journal:  Circulation       Date:  2005-01-17       Impact factor: 29.690

4.  A mutation in the Z-line Cypher/ZASP protein is associated with arrhythmogenic right ventricular cardiomyopathy.

Authors:  J M Lopez-Ayala; M Ortiz-Genga; I Gomez-Milanes; D Lopez-Cuenca; F Ruiz-Espejo; J J Sanchez-Munoz; M J Oliva-Sandoval; L Monserrat; J R Gimeno
Journal:  Clin Genet       Date:  2014-09-08       Impact factor: 4.438

5.  Long-term follow-up of patients with short QT syndrome.

Authors:  Carla Giustetto; Rainer Schimpf; Andrea Mazzanti; Chiara Scrocco; Philippe Maury; Olli Anttonen; Vincent Probst; Jean-Jacques Blanc; Pascal Sbragia; Paola Dalmasso; Martin Borggrefe; Fiorenzo Gaita
Journal:  J Am Coll Cardiol       Date:  2011-08-02       Impact factor: 24.094

Review 6.  Short QT syndrome.

Authors:  Rainer Schimpf; Christian Wolpert; Fiorenzo Gaita; Carla Giustetto; Martin Borggrefe
Journal:  Cardiovasc Res       Date:  2005-08-15       Impact factor: 10.787

7.  Increase in sudden death from coronary artery disease in young adults.

Authors:  Dabit Arzamendi; Begoña Benito; Helena Tizon-Marcos; Jose Flores; Jean François Tanguay; Hung Ly; Serge Doucet; Louis Leduc; Tack Ki Leung; Oscar Campuzano; Anna Iglesias; Mario Talajic; Ramon Brugada
Journal:  Am Heart J       Date:  2011-03       Impact factor: 4.749

8.  Primary carnitine deficiency: novel mutations and insights into the cardiac phenotype.

Authors:  K Shibbani; A C Fahed; L Al-Shaar; M Arabi; G Nemer; F Bitar; M Majdalani
Journal:  Clin Genet       Date:  2013-03-12       Impact factor: 4.438

9.  FLNC Gene Splice Mutations Cause Dilated Cardiomyopathy.

Authors:  Rene L Begay; Charles A Tharp; August Martin; Sharon L Graw; Gianfranco Sinagra; Daniela Miani; Mary E Sweet; Dobromir B Slavov; Neil Stafford; Molly J Zeller; Rasha Alnefaie; Teisha J Rowland; Francesca Brun; Kenneth L Jones; Katherine Gowan; Luisa Mestroni; Deborah M Garrity; Matthew R G Taylor
Journal:  JACC Basic Transl Sci       Date:  2016-07-27

10.  The genetic component of Brugada syndrome.

Authors:  Morten W Nielsen; Anders G Holst; Søren-Peter Olesen; Morten S Olesen
Journal:  Front Physiol       Date:  2013-07-15       Impact factor: 4.566

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.