Literature DB >> 31472821

Molecular Autopsy Implicates Primary Carnitine Deficiency in Sudden Unexplained Death and Reversible Short QT Syndrome.

Roselle Gélinas1, Emma Leach2, Gabriella Horvath3, Zachary Laksman4.   

Abstract

We report a case of sudden unexplained death in a young asymptomatic woman in whom postmortem genetic testing after a negative autopsy identified a homozygous pathogenic mutation in SLC22A5 which leads clinically to primary carnitine deficiency (PCD). Her brother was subsequently diagnosed clinically with short QT syndrome, received an implantable defibrillator, and was then found to carry the same pathogenic homozygous mutation and critically low levels of carnitine. His QT interval improved with the use of carnitine supplementation, highlighting the close relationship between electrophysiology and biochemistry, and the importance of postmortem genetic testing in the clinical management of surviving relatives.
Copyright © 2019 Canadian Cardiovascular Society. Published by Elsevier Inc. All rights reserved.

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Year:  2019        PMID: 31472821     DOI: 10.1016/j.cjca.2019.05.014

Source DB:  PubMed          Journal:  Can J Cardiol        ISSN: 0828-282X            Impact factor:   5.223


  7 in total

1.  European Heart Rhythm Association (EHRA)/Heart Rhythm Society (HRS)/Asia Pacific Heart Rhythm Society (APHRS)/Latin American Heart Rhythm Society (LAHRS) Expert Consensus Statement on the state of genetic testing for cardiac diseases.

Authors:  Arthur A M Wilde; Christopher Semsarian; Manlio F Márquez; Alireza Sepehri Shamloo; Michael J Ackerman; Euan A Ashley; Back Sternick Eduardo; Héctor Barajas-Martinez; Elijah R Behr; Connie R Bezzina; Jeroen Breckpot; Philippe Charron; Priya Chockalingam; Lia Crotti; Michael H Gollob; Steven Lubitz; Naomasa Makita; Seiko Ohno; Martín Ortiz-Genga; Luciana Sacilotto; Eric Schulze-Bahr; Wataru Shimizu; Nona Sotoodehnia; Rafik Tadros; James S Ware; David S Winlaw; Elizabeth S Kaufman; Takeshi Aiba; Andreas Bollmann; Jong-Il Choi; Aarti Dalal; Francisco Darrieux; John Giudicessi; Mariana Guerchicoff; Kui Hong; Andrew D Krahn; Ciorsti Mac Intyre; Judith A Mackall; Lluís Mont; Carlo Napolitano; Pablo Ochoa Juan; Petr Peichl; Alexandre C Pereira; Peter J Schwartz; Jon Skinner; Christoph Stellbrink; Jacob Tfelt-Hansen; Thomas Deneke
Journal:  J Arrhythm       Date:  2022-05-31

2.  European Heart Rhythm Association (EHRA)/Heart Rhythm Society (HRS)/Asia Pacific Heart Rhythm Society (APHRS)/Latin American Heart Rhythm Society (LAHRS) Expert Consensus Statement on the state of genetic testing for cardiac diseases.

Authors:  Arthur A M Wilde; Christopher Semsarian; Manlio F Márquez; Alireza Sepehri Shamloo; Michael J Ackerman; Euan A Ashley; Eduardo Back Sternick; Héctor Barajas-Martinez; Elijah R Behr; Connie R Bezzina; Jeroen Breckpot; Philippe Charron; Priya Chockalingam; Lia Crotti; Michael H Gollob; Steven Lubitz; Naomasa Makita; Seiko Ohno; Martín Ortiz-Genga; Luciana Sacilotto; Eric Schulze-Bahr; Wataru Shimizu; Nona Sotoodehnia; Rafik Tadros; James S Ware; David S Winlaw; Elizabeth S Kaufman; Takeshi Aiba; Andreas Bollmann; Jong Il Choi; Aarti Dalal; Francisco Darrieux; John Giudicessi; Mariana Guerchicoff; Kui Hong; Andrew D Krahn; Ciorsti MacIntyre; Judith A Mackall; Lluís Mont; Carlo Napolitano; Juan Pablo Ochoa; Petr Peichl; Alexandre C Pereira; Peter J Schwartz; Jon Skinner; Christoph Stellbrink; Jacob Tfelt-Hansen; Thomas Deneke
Journal:  Europace       Date:  2022-09-01       Impact factor: 5.486

3.  Evaluation of gene validity for CPVT and short QT syndrome in sudden arrhythmic death.

Authors:  Roddy Walsh; Arnon Adler; Ahmad S Amin; Emanuela Abiusi; Melanie Care; Hennie Bikker; Simona Amenta; Harriet Feilotter; Eline A Nannenberg; Francesco Mazzarotto; Valentina Trevisan; John Garcia; Ray E Hershberger; Marco V Perez; Amy C Sturm; James S Ware; Wojciech Zareba; Valeria Novelli; Arthur A M Wilde; Michael H Gollob
Journal:  Eur Heart J       Date:  2022-04-14       Impact factor: 35.855

4.  Primary carnitine deficiency - diagnosis after heart transplantation: better late than never!

Authors:  Sarah C Grünert; Sara Tucci; Anke Schumann; Meike Schwendt; Gwendolyn Gramer; Georg F Hoffmann; Michelle Erbel; Brigitte Stiller; Ute Spiekerkoetter
Journal:  Orphanet J Rare Dis       Date:  2020-04-10       Impact factor: 4.123

5.  Increased detection of primary carnitine deficiency through second-tier newborn genetic screening.

Authors:  Yiming Lin; Weifeng Zhang; Chenggang Huang; Chunmei Lin; Weihua Lin; Weilin Peng; Qingliu Fu; Dongmei Chen
Journal:  Orphanet J Rare Dis       Date:  2021-03-23       Impact factor: 4.123

Review 6.  Clinical characteristics of primary carnitine deficiency: A structured review using a case-by-case approach.

Authors:  Loek L Crefcoeur; Gepke Visser; Sacha Ferdinandusse; Frits A Wijburg; Mirjam Langeveld; Barbara Sjouke
Journal:  J Inherit Metab Dis       Date:  2022-02-03       Impact factor: 4.750

Review 7.  Clinical Genetics of Inherited Arrhythmogenic Disease in the Pediatric Population.

Authors:  Estefanía Martínez-Barrios; Sergi Cesar; José Cruzalegui; Clara Hernandez; Elena Arbelo; Victoria Fiol; Josep Brugada; Ramon Brugada; Oscar Campuzano; Georgia Sarquella-Brugada
Journal:  Biomedicines       Date:  2022-01-05
  7 in total

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