Literature DB >> 27539165

The natural history of fetal long QT syndrome.

Bettina F Cuneo1, Janette F Strasburger2, Ronald T Wakai3.   

Abstract

INTRODUCTION: Fetal magnetocardiography (fMCG), the magnetic analog of ECG, has provided invaluable insight into the mechanisms of fetal arrhythmias. In the past 15years, we have evaluated over 300 fetuses with arrhythmia by fMCG. We review the unique characteristics and natural history of the long QT syndrome (LQTS) rhythms.
METHODS: We reviewed the fMCGs of subjects referred with suspected LQTS based on either a positive family history or echo diagnosis of the LQTS rhythms (sinus bradycardia, ventricular tachycardia, or 2:1 AV conduction) to the Biomagnetism laboratory in the Department of Medical Physics, UW-Madison. We recorded fMCGs using a 37-channel (Magnes, 4D Neuroimaging, Inc., San Diego, CA) superconducting quantum interference device (SQUID) biomagnetometer, housed in a magnetically-shielded room for 1200-6000s. Signal processing was used to remove maternal interference. Cardiac intervals (R-R, p, QRS, QT) were measured and compared to published normals. We correlated fetal heart rate (FHR) patterns and effects of fetal movement on FHR and rhythm using actocardiography.
RESULTS: Thirty-nine fetuses were studied at a mean of 28 (19-38) weeks of gestation. All had structurally normal hearts. One was on amiodarone for suspected supraventricular tachycardia and hydrops. Five had serial fMCGs. Isolated sinus bradycardia with a QTc >490ms was found in 35: 33 had a KCNQ1 mutation There was one false positive and one false negative LQTS diagnosis. Four fetuses had torsades de pointes (TdP) and 3 had periods of 2:1 conduction and either KCNH2 or SCN5A mutations. TdP was rarely initiated with a preceding long-short pattern and did not degenerate into ventricular fibrillation. One fetus with TdP died in utero, 2 with fetal TdP had postnatal cardiac arrest.
CONCLUSION: Fetal LQTS is diagnosed by an fMCG QTc >490ms with an 89% sensitivity and specificity. TdP are seen with uncharacterized, KCNH2 or SCN5A R1623q mutations. Fetal TdP occurs when QTc ≥620ms. Identifying fetal LQTS and defining its rhythms by fMCG risk stratifies postnatal management.
Copyright © 2016 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Arrhythmia; Fetus; KCNH2 mutation; Long QT syndrome; Torsades de pointes

Mesh:

Year:  2016        PMID: 27539165      PMCID: PMC5159227          DOI: 10.1016/j.jelectrocard.2016.07.023

Source DB:  PubMed          Journal:  J Electrocardiol        ISSN: 0022-0736            Impact factor:   1.438


  28 in total

1.  Stillbirths, sudden infant deaths, and long-QT syndrome: puzzle or mosaic, the pieces of the Jigsaw are being fitted together.

Authors:  Peter J Schwartz
Journal:  Circulation       Date:  2004-06-22       Impact factor: 29.690

2.  Cardiotocography plus ST analysis of fetal electrocardiogram compared with cardiotocography only for intrapartum monitoring: a randomized controlled trial.

Authors:  Michelle E M H Westerhuis; Gerard H A Visser; Karel G M Moons; Erik van Beek; Manon J Benders; Saskia M Bijvoet; Hendrikus J H M van Dessel; Addy P Drogtrop; Herman P van Geijn; Giuseppe C Graziosi; Floris Groenendaal; Jan M M van Lith; Jan G Nijhuis; S Guid Oei; Herman P Oosterbaan; Martina M Porath; Robbert J P Rijnders; Nico W E Schuitemaker; Louisa M Sopacua; Ingeborg van der Tweel; Lia D E Wijnberger; Christine Willekes; Nicolaas P A Zuithoff; Ben Willem J Mol; Anneke Kwee
Journal:  Obstet Gynecol       Date:  2010-06       Impact factor: 7.661

3.  Arrhythmia phenotype during fetal life suggests long-QT syndrome genotype: risk stratification of perinatal long-QT syndrome.

Authors:  Bettina F Cuneo; Susan P Etheridge; Hitoshi Horigome; Denver Sallee; Anita Moon-Grady; Hsin-Yi Weng; Michael J Ackerman; D Woodrow Benson
Journal:  Circ Arrhythm Electrophysiol       Date:  2013-08-30

4.  ACOG Practice Bulletin No. 106: Intrapartum fetal heart rate monitoring: nomenclature, interpretation, and general management principles.

Authors: 
Journal:  Obstet Gynecol       Date:  2009-07       Impact factor: 7.661

5.  Prenatal diagnosis of long QT syndrome: implications for delivery room and neonatal management.

Authors:  E Anne Greene; Charles I Berul; Mary T Donofrio
Journal:  Cardiol Young       Date:  2012-05-02       Impact factor: 1.093

6.  Third trimester fetal heart rate predicts phenotype and mutation burden in the type 1 long QT syndrome.

Authors:  Annika Winbo; Inger Fosdal; Maria Lindh; Ulla-Britt Diamant; Johan Persson; Göran Wettrell; Annika Rydberg
Journal:  Circ Arrhythm Electrophysiol       Date:  2015-05-27

7.  Calmodulin mutations associated with recurrent cardiac arrest in infants.

Authors:  Lia Crotti; Christopher N Johnson; Elisabeth Graf; Gaetano M De Ferrari; Bettina F Cuneo; Marc Ovadia; John Papagiannis; Michael D Feldkamp; Subodh G Rathi; Jennifer D Kunic; Matteo Pedrazzini; Thomas Wieland; Peter Lichtner; Britt-Maria Beckmann; Travis Clark; Christian Shaffer; D Woodrow Benson; Stefan Kääb; Thomas Meitinger; Tim M Strom; Walter J Chazin; Peter J Schwartz; Alfred L George
Journal:  Circulation       Date:  2013-02-06       Impact factor: 29.690

8.  Electrophysiological characteristics of fetal atrioventricular block.

Authors:  Hui Zhao; Bettina F Cuneo; Janette F Strasburger; James C Huhta; Nina L Gotteiner; Ronald T Wakai
Journal:  J Am Coll Cardiol       Date:  2008-01-01       Impact factor: 24.094

9.  The long QT syndrome: a prospective international study.

Authors:  A J Moss; P J Schwartz; R S Crampton; E Locati; E Carleen
Journal:  Circulation       Date:  1985-01       Impact factor: 29.690

10.  Electrophysiologic features of fetal ventricular aneurysms and diverticula.

Authors:  Carli Peters; Annette Wacker-Gussmann; Janette F Strasburger; Bettina F Cuneo; Nina L Gotteiner; Mehemet Gulecyuz; Ronald T Wakai
Journal:  Prenat Diagn       Date:  2014-12-19       Impact factor: 3.050

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  5 in total

1.  Fetal diagnosis of KCNQ1-variant long QT syndrome using fetal echocardiography and magnetocardiography.

Authors:  Lajja Desai; Ron Wakai; Sabrina Tsao; Janette Strasburger; Nina Gotteiner; Angira Patel
Journal:  Pacing Clin Electrophysiol       Date:  2020-04-07       Impact factor: 1.976

2.  Noninvasive Fetal Electrocardiography in the Diagnosis of Long QT Syndrome: A Case Series.

Authors:  Neeta Sethi; Kiyoe Funamoto; Catherine Ingbar; Paige Mass; Jeffrey Moak; Ronald Wakai; Janette Strasburger; Mary Donofrio; Ahsan Khandoker; Yoshitaka Kimura; Anita Krishnan
Journal:  Fetal Diagn Ther       Date:  2020-07-02       Impact factor: 2.587

3.  A Potential Diagnostic Approach for Foetal Long-QT Syndrome, Developed and Validated in Children.

Authors:  Arja Suzanne Vink; Irene M Kuipers; Rianne H A C M De Bruin-Bon; Arthur A M Wilde; Nico A Blom; Sally-Ann B Clur
Journal:  Pediatr Cardiol       Date:  2018-05-22       Impact factor: 1.655

4.  Maternal mosaicism in long QT syndrome due to a pathogenic variant in KCNH2.

Authors:  Briana L Sawyer; Martin Tristani-Firouzi; Layne E Wells; Matteo Vatta; Susan P Etheridge
Journal:  HeartRhythm Case Rep       Date:  2020-11-16

5.  Magnetocardiography on an isolated animal heart with a room-temperature optically pumped magnetometer.

Authors:  Kasper Jensen; Mark Alexander Skarsfeldt; Hans Stærkind; Jens Arnbak; Mikhail V Balabas; Søren-Peter Olesen; Bo Hjorth Bentzen; Eugene S Polzik
Journal:  Sci Rep       Date:  2018-11-01       Impact factor: 4.379

  5 in total

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