NKX2-5 genetic mutation in a young woman with an atrial septal defect presenting with complete heart block: ICD or bradycardia pacemaker?

被引:0
作者
El-Medany, Ahmed [1 ,2 ]
Aziz, Shahid [2 ]
Duncan, Edward [1 ]
机构
[1] Bristol Heart Inst, Cardiol, Bristol, England
[2] Southmead Hosp, Cardiol, Bristol, England
关键词
Pacing and electrophysiology; Arrhythmias; Genetic screening; counselling; CARDIOMYOPATHY; DISEASE; DEATH;
D O I
10.1136/bcr-2022-252523
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
A woman in her 40s was admitted following syncope. The 12-lead ECG showed atrial fibrillation with slow ventricular response and suspected complete atrioventricular (AV) block. Cardiac monitoring demonstrated non-sustained monomorphic ventricular tachycardia (VT). Her medical history included surgical repair of an atrial septal defect (ASD) aged 4 years. The patient's mother died suddenly in her early 50s and also had an ASD. Given the patient's syncope, background of familial sudden cardiac death (SCD), suspicion of complete AV block and non-sustained VT, she received an implantable cardiac defibrillator (ICD). She underwent genetic testing, revealing a heterozygous NKX2-5 genetic mutation. The signature phenotype in NKX2-5 mutations is ASD with AV conduction disturbance and an increased risk of SCD secondary to ventricular arrhythmias or severe bradycardia. SCD has been described in NKX2-5 mutation carriers despite functioning permanent pacemakers (PPMs). Therefore, we propose implantation of a preventive ICD, as opposed to a PPM.
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页数:4
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共 14 条
[1]   NKX2-5 regulates human cardiomyogenesis via a HEY2 dependent transcriptional network [J].
Anderson, David J. ;
Kaplan, David I. ;
Bell, Katrina M. ;
Koutsis, Katerina ;
Haynes, John M. ;
Mills, Richard J. ;
Phelan, Dean G. ;
Qian, Elizabeth L. ;
Leitoguinho, Ana Rita ;
Arasaratnam, Deevina ;
Labonne, Tanya ;
Ng, Elizabeth S. ;
Davis, Richard P. ;
Casini, Simona ;
Passier, Robert ;
Hudson, James E. ;
Porrello, Enzo R. ;
Costa, Mauro W. ;
Rafii, Arash ;
Curl, Clare L. ;
Delbridge, Lea M. ;
Harvey, Richard P. ;
Oshlack, Alicia ;
Cheung, Michael M. ;
Mummery, Christine L. ;
Petrou, Stephen ;
Elefanty, Andrew G. ;
Stanley, Edouard G. ;
Elliott, David A. .
NATURE COMMUNICATIONS, 2018, 9
[2]  
Baumgartner Helmut, 2021, Eur Heart J, V42, P563, DOI [10.1093/eurheartj/ehaa554, 10.15829/1560-4071-2021-4702]
[3]   Genetics of Congenital Heart Defects: The NKX2-5 Gene, a Key Player [J].
Chung, Ill-Min ;
Rajakumar, Govindasamy .
GENES, 2016, 7 (02)
[4]   Familial Atrial Septal Defect and Sudden Cardiac Death: Identification of a Novel NKX2-5 Mutation and a Review of the Literature [J].
Ellesoe, Sabrina Gade ;
Johansen, Morten Munk ;
Bjerre, Jesper Vandborg ;
Hjortdal, Vibeke Elisabeth ;
Brunak, Soren ;
Larsen, Lars Allan .
CONGENITAL HEART DISEASE, 2016, 11 (03) :283-290
[5]   A comprehensive in silico analysis, distribution and frequency of human Nkx2-5 mutations; A critical gene in congenital heart disease [J].
Kalayinia, Samira ;
Ghasemi, Serwa ;
Mahdieh, Nejat .
JOURNAL OF CARDIOVASCULAR AND THORACIC RESEARCH, 2019, 11 (04) :287-299
[6]   An update on genetic variants of the NKX2-5 [J].
Kolomenski, Jorge E. ;
Delea, Marisol ;
Simonetti, Leandro ;
Fabbro, Monica C. ;
Espeche, Lucia D. ;
Taboas, Melisa ;
Nadra, Alejandro D. ;
Bruque, Carlos D. ;
Dain, Liliana .
HUMAN MUTATION, 2020, 41 (07) :1187-1208
[7]   Cardiac Phenotype and Long-Term Follow-Up of Patients With Mutations in NKX2-5 Gene [J].
Maury, Philippe ;
Gandjbakhch, Estelle ;
Baruteau, Alban-Elouen ;
Bessiere, Francis ;
Kyndt, Florence ;
Bouvagnet, Patrice ;
Rollin, Anne ;
Bonnet, Damien ;
Probst, Vincent ;
Maltret, Alice .
JOURNAL OF THE AMERICAN COLLEGE OF CARDIOLOGY, 2016, 68 (21) :2389-2390
[8]   NKX2.5 mutations in patients with congenital heart disease [J].
McElhinney, DB ;
Geiger, E ;
Blinder, J ;
Benson, DW ;
Goldmuntz, E .
JOURNAL OF THE AMERICAN COLLEGE OF CARDIOLOGY, 2003, 42 (09) :1650-1655
[9]   Case Report: A Novel NKX2-5 Mutation in a Family With Congenital Heart Defects, Left Ventricular Non-compaction, Conduction Disease, and Sudden Cardiac Death [J].
Morlanes-Gracia, Paula ;
Antoniutti, Guido ;
Alvarez-Rubio, Jorge ;
Torres-Juan, Laura ;
Heine-Suner, Damian ;
Ripoll-Vera, Tomas .
FRONTIERS IN CARDIOVASCULAR MEDICINE, 2021, 8
[10]   Nkx2-5 pathways and congenital heart disease: Loss of ventricular myocyte lineage progressive cardiomyopathy and complete heart block [J].
Pashmforoush, M ;
Lu, JT ;
Chen, HY ;
St Amand, T ;
Kondo, R ;
Pradervand, S ;
Evans, SM ;
Clark, B ;
Feramisco, JR ;
Giles, W ;
Ho, SY ;
Benson, DW ;
Silberbach, M ;
Shou, WN ;
Chien, KR .
CELL, 2004, 117 (03) :373-386