Literature DB >> 28699125

Uptake of Predictive Genetic Testing and Cardiac Evaluation for Children at Risk for an Inherited Arrhythmia or Cardiomyopathy.

Susan Christian1, Joseph Atallah2, Robin Clegg3, Michael Giuffre3, Cathleen Huculak4, Tara Dzwiniel5, Jillian Parboosingh4, Sherryl Taylor5, Martin Somerville5.   

Abstract

Predictive genetic testing in minors should be considered when clinical intervention is available. Children who carry a pathogenic variant for an inherited arrhythmia or cardiomyopathy require regular cardiac screening and may be prescribed medication and/or be told to modify their physical activity. Medical genetics and pediatric cardiology charts were reviewed to identify factors associated with uptake of genetic testing and cardiac evaluation for children at risk for long QT syndrome, hypertrophic cardiomyopathy or arrhythmogenic right ventricular cardiomyopathy. The data collected included genetic diagnosis, clinical symptoms in the carrier parent, number of children under 18 years of age, age of children, family history of sudden cardiac arrest/death, uptake of cardiac evaluation and if evaluated, phenotype for each child. We identified 97 at risk children from 58 families found to carry a pathogenic variant for one of these conditions. Sixty six percent of the families pursued genetic testing and 73% underwent cardiac screening when it was recommended. Declining predictive genetic testing was significantly associated with genetic specialist recommendation (p < 0.001) and having an asymptomatic carrier father (p = 0.006). Cardiac evaluation was significantly associated with uptake of genetic testing (p = 0.007). This study provides a greater understanding of factors associated with uptake of genetic testing and cardiac evaluation in children at risk of an inherited arrhythmia or cardiomyopathy. It also identifies a need to educate families about the importance of cardiac evaluation even in the absence of genetic testing.

Entities:  

Keywords:  Arrhythmogenic right ventricular cardiomyopathy; Children; Genetic testing; Hypertrophic cardiomyopathy; Long QT syndrome; Predictive

Mesh:

Year:  2017        PMID: 28699125     DOI: 10.1007/s10897-017-0129-0

Source DB:  PubMed          Journal:  J Genet Couns        ISSN: 1059-7700            Impact factor:   2.537


  15 in total

1.  Uptake of genetic testing for cancer predisposition.

Authors:  D G Evans; E R Maher; R Macleod; D R Davies; D Craufurd
Journal:  J Med Genet       Date:  1997-09       Impact factor: 6.318

2.  A population-based survey in Australia of men's and women's perceptions of genetic risk and predictive genetic testing and implications for primary care.

Authors:  S Taylor
Journal:  Public Health Genomics       Date:  2011-04-13       Impact factor: 2.000

3.  Factors Associated with Uptake of Genetics Services for Hypertrophic Cardiomyopathy.

Authors:  Amirah Khouzam; Andrea Kwan; Samantha Baxter; Jonathan A Bernstein
Journal:  J Genet Couns       Date:  2015-01-08       Impact factor: 2.537

4.  Constructing "best interests": genetic testing of children in families with hypertrophic cardiomyopathy.

Authors:  Els Geelen; Ine Van Hoyweghen; Pieter A Doevendans; Carlo L M Marcelis; Klasien Horstman
Journal:  Am J Med Genet A       Date:  2011-07-07       Impact factor: 2.802

Review 5.  Points to Consider: Ethical, Legal, and Psychosocial Implications of Genetic Testing in Children and Adolescents.

Authors:  Jeffrey R Botkin; John W Belmont; Jonathan S Berg; Benjamin E Berkman; Yvonne Bombard; Ingrid A Holm; Howard P Levy; Kelly E Ormond; Howard M Saal; Nancy B Spinner; Benjamin S Wilfond; Joseph D McInerney
Journal:  Am J Hum Genet       Date:  2015-07-02       Impact factor: 11.025

Review 6.  Recommendations for the use of genetic testing in the clinical evaluation of inherited cardiac arrhythmias associated with sudden cardiac death: Canadian Cardiovascular Society/Canadian Heart Rhythm Society joint position paper.

Authors:  Michael H Gollob; Louis Blier; Ramon Brugada; Jean Champagne; Vijay Chauhan; Sean Connors; Martin Gardner; Martin S Green; Robert Gow; Robert Hamilton; Louise Harris; Jeff S Healey; Kathleen Hodgkinson; Christina Honeywell; Michael Kantoch; Joel Kirsh; Andrew Krahn; Michelle Mullen; Ratika Parkash; Damian Redfearn; Julie Rutberg; Shubhayan Sanatani; Anna Woo
Journal:  Can J Cardiol       Date:  2011 Mar-Apr       Impact factor: 5.223

7.  Parental attitudes, beliefs, and perceptions about genetic testing for FAP and colorectal cancer surveillance in minors.

Authors:  Fallon R Levine; James E Coxworth; David A Stevenson; Thérèse Tuohy; Randall W Burt; Anita Y Kinney
Journal:  J Genet Couns       Date:  2010-03-02       Impact factor: 2.537

8.  Uptake of genetic counselling and predictive DNA testing in hypertrophic cardiomyopathy.

Authors:  Imke Christiaans; Erwin Birnie; Gouke J Bonsel; Arthur Am Wilde; Irene M van Langen
Journal:  Eur J Hum Genet       Date:  2008-05-14       Impact factor: 4.246

9.  Pre-symptomatic genetic testing for inherited cardiac conditions: a qualitative exploration of psychosocial and ethical implications.

Authors:  Elizabeth Ormondroyd; Stephanie Oates; Michael Parker; Edward Blair; Hugh Watkins
Journal:  Eur J Hum Genet       Date:  2013-05-01       Impact factor: 4.246

10.  Feasibility of an Assessment Tool for Children's Competence to Consent to Predictive Genetic Testing: a Pilot Study.

Authors:  Irma M Hein; Pieter W Troost; Robert Lindeboom; Imke Christiaans; Thomas Grisso; Johannes B van Goudoever; Ramón J L Lindauer
Journal:  J Genet Couns       Date:  2015-04-26       Impact factor: 2.537

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  5 in total

1.  Managing uncertainty in inherited cardiac pathologies-an international multidisciplinary survey.

Authors:  Terri Patricia McVeigh; Luke J Kelly; Elizabeth Whitmore; Tara Clark; Brendan Mullaney; David E Barton; Alana Ward; Sally Ann Lynch
Journal:  Eur J Hum Genet       Date:  2019-04-12       Impact factor: 4.246

Review 2.  Family Communication About Genetic Risk of Hereditary Cardiomyopathies and Arrhythmias: an Integrative Review.

Authors:  Lisa L Shah; Sandra Daack-Hirsch
Journal:  J Genet Couns       Date:  2018-02-28       Impact factor: 2.537

3.  2020 APHRS/HRS expert consensus statement on the investigation of decedents with sudden unexplained death and patients with sudden cardiac arrest, and of their families.

Authors:  Martin K Stiles; Arthur A M Wilde; Dominic J Abrams; Michael J Ackerman; Christine M Albert; Elijah R Behr; Sumeet S Chugh; Martina C Cornel; Karen Gardner; Jodie Ingles; Cynthia A James; Jyh-Ming Jimmy Juang; Stefan Kääb; Elizabeth S Kaufman; Andrew D Krahn; Steven A Lubitz; Heather MacLeod; Carlos A Morillo; Koonlawee Nademanee; Vincent Probst; Elizabeth V Saarel; Luciana Sacilotto; Christopher Semsarian; Mary N Sheppard; Wataru Shimizu; Jonathan R Skinner; Jacob Tfelt-Hansen; Dao Wu Wang
Journal:  Heart Rhythm       Date:  2020-10-19       Impact factor: 6.343

4.  2020 APHRS/HRS expert consensus statement on the investigation of decedents with sudden unexplained death and patients with sudden cardiac arrest, and of their families.

Authors:  Martin K Stiles; Arthur A M Wilde; Dominic J Abrams; Michael J Ackerman; Christine M Albert; Elijah R Behr; Sumeet S Chugh; Martina C Cornel; Karen Gardner; Jodie Ingles; Cynthia A James; Jyh-Ming Jimmy Juang; Stefan Kääb; Elizabeth S Kaufman; Andrew D Krahn; Steven A Lubitz; Heather MacLeod; Carlos A Morillo; Koonlawee Nademanee; Vincent Probst; Elizabeth V Saarel; Luciana Sacilotto; Christopher Semsarian; Mary N Sheppard; Wataru Shimizu; Jonathan R Skinner; Jacob Tfelt-Hansen; Dao Wu Wang
Journal:  J Arrhythm       Date:  2021-04-08

Review 5.  Cascade health service use in family members following genetic testing in children: a scoping literature review.

Authors:  Alexandra Cernat; Robin Z Hayeems; Wendy J Ungar
Journal:  Eur J Hum Genet       Date:  2021-08-26       Impact factor: 4.246

  5 in total

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