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Complex and Novel Arrhythmias Precede Stillbirth in Fetuses With De Novo Long QT Syndrome
Open accessAbstract
Background: Long QT syndrome (LQTS) is a leading cause of sudden cardiac death in early life and has been implicated in ≈10% of sudden infant deaths and unexplained stillbirths. The purpose of our study was to use fetal magnetocardiography to characterize the electrophysiology and rhythm phenotypes of fetuses with de novo and inherited LQTS variants and identify risk factors for sudden death before birth. Methods: We reviewed the fetal magnetocardiography database from the University of Wisconsin Biomagnetism Laboratory for fetuses with confirmed LQTS. We assessed waveform intervals, heart rate, and rhythm, including the signature LQTS rhythms: functional 2° atrioventricular block, T-wave alternans, and torsade de pointes (TdP). Results: Thirty-nine fetuses had pathogenic variants in LQTS genes: 27 carried the family variant, 11 had de novo variants, and 1 was indeterminate. De novo variants, especially de novo SCN5A variants, were strongly associated with a severe rhythm phenotype and perinatal death: 9 (82%) showed signature LQTS rhythms, 6 (55%) showed TdP, 5 (45%) were stillborn, and 1 (9%) died in infancy. Those that died exhibited novel fetal rhythms, including atrioventricular block with 3:1 conduction ratio, QRS alternans in 2:1 atrioventricular block, long-cycle length TdP, and slow monomorphic ventricular tachycardia. Premature ventricular contractions were also strongly associated with TdP and perinatal death. Fetuses with familial variants showed a lower incidence of signature LQTS rhythm (6/27=22%), including TdP (3/27=11%). All were live born. Conclusions: The malignancy of de novo LQTS variants was remarkably high and demonstrate that these mutations are a significant cause of stillbirth. Their ability to manifest rhythms not known to be associated with LQTS increases the difficulty of echocardiographic diagnosis and decreases the likelihood that a resultant fetal loss is attributed to LQTS. Registration: URL: http://www.clinicaltrials.gov . Unique identifier: NCT03047161.
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OPMs have improved miniaturization making them wearable, though they still require heavy EMS.
“More recently, OPMs have improved the level of miniaturization making them even wearable, opening additional avenues for a more flexible use in multiple clinical applications (270, 273, 274), despite still requiring heavy EMS.”
From Clinical magnetocardiography: the unshielded bet—past, present, and future (2023) · cites this paper as [274]
The Methods (Data acquisition; page unavailable) describe SQUID magnetometers housed in a magnetically shielded room, not wearable OPMs. The Conclusions mention a cost-effective fetal magnetocardiography system, but our copy does not address OPM miniaturization, wearability, or whether OPMs require heavy electromagnetic shielding (searched: OPM, wearable, shielding). (LLM, conf=0.76)
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Metadata
- Authors
- Strand, S., Strasburger, J. F., Cuneo, B. F., & Wakai, R. T.
- Affiliations
- University of Wisconsin–Madison (US)
- Children's Hospital of Wisconsin (US)
- Children's Hospital Colorado (US)
- Corresponding author
- Wakai, Ronald T.
- Venue
- Circulation: Arrhythmia and Electrophysiology
- Published
- 2020-05
- Volume
- 13
- Issue
- 5
- Pages
- e008082-e008082
- ISSN
- 1941-3084 (ISSN-L)
- 1941-3149
- PMC
- PMC7241276
- OpenAlex
- W3028362556
- Cited by
- 50
- References
- 14
- Citations by year
- 2026: 7 · 2025: 7 · 2024: 8 · 2023: 10 · 2022: 10 · 2021: 4 · 2020: 4
- FWCI
- 4.33
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Circulation Arrhythmia and Electrophysiology (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- Funders
- National Institutes of Health
- National Heart, Lung, and Blood Institute (R01HL143485, R01HL063174)
- Topics
- Health Sciences › Medicine › Cardiology and Cardiovascular Medicine › Cardiac electrophysiology and arrhythmias
- Health Sciences › Medicine › Cardiology and Cardiovascular Medicine › Cardiovascular Effects of Exercise
- Health Sciences › Medicine › Cardiology and Cardiovascular Medicine › Cardiac pacing and defibrillation studies
- Keywords
- long QT syndrome, de novo mutations, fetal magnetocardiography, stillbirth, perinatal death, torsades de pointes, T-wave alternans, atrioventricular block, SCN5A, fetal arrhythmia, sudden cardiac death, ventricular tachycardia, premature ventricular contractions, QRS alternans
- MeSH terms
- Cause of Death, Female, Fetal Heart, Gestational Age, Heart Rate, Fetal, Humans, Long QT Syndrome, Mutation, Phenotype, Predictive Value of Tests, Pregnancy, Prenatal Diagnosis, Risk Factors, Databases, Factual, Risk Assessment, Genetic Predisposition to Disease, Heredity, Stillbirth, Magnetocardiography
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