Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2022; 10(19): 6602-6608
Published online Jul 6, 2022. doi: 10.12998/wjcc.v10.i19.6602
Unusual course of congenital complete heart block in an adult: A case report
Li-Na Su, Man-Yan Wu, Yu-Xia Cui, Chong-You Lee, Jun-Xian Song, Hong Chen
Li-Na Su, Man-Yan Wu, Yu-Xia Cui, Chong-You Lee, Jun-Xian Song, Hong Chen, Department of Cardiology, Peking University People’s Hospital, Beijing 100044, China
Author contributions: Su LN and Wu MY reviewed the literature and contributed to manuscript drafting; Cui YX extracted the data and collected the clinical information; Lee CY and Song JX analyzed and interpreted the imaging findings; Chen H revised the manuscript; all authors issued final approval for the version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Hong Chen, MD, PhD, Professor, Department of Cardiology, Peking University People’s Hospital, No. 11 Xizhimen South Street, Beijing 100044, China. chenhongbj@medmail.com.cn
Received: December 1, 2021
Peer-review started: December 1, 2021
First decision: January 12, 2022
Revised: January 20, 2022
Accepted: May 13, 2022
Article in press: May 13, 2022
Published online: July 6, 2022
Processing time: 204 Days and 22.4 Hours
Abstract
BACKGROUND

Congenital complete heart block (CCHB) with normal cardiac structure and negativity for anti-Ro/La antibody is rare. Additionally, CCHB is much less frequently diagnosed in adults, and its natural history in adults is less well known.

CASE SUMMARY

A 23-year-old woman was admitted to our hospital for frequent syncopal episodes. She had bradycardia at the age of 1 year but had never had impaired exercise capacity or a syncopal episode before admission. The possible diagnosis of acquired complete atrioventricular block was carefully ruled out, and then the diagnosis of CCHB was made. According to existing guidelines, permanent pacemaker implantation was recommended, but the patient declined. With regular follow-up for 28 years, the patient had an unusually good outcome without any invasive intervention or medicine. She had an uneventful pregnancy and led a normally active life without any symptoms of low cardiac output or syncopal recurrence.

CONCLUSION

This case implies that CCHB in adulthood may have good clinical outcomes and does not always require permanent pacemaker implantation.

Keywords: Congenital complete heart block; Acquired complete atrioventricular block; Syncope; Pacemaker implantation; Case report

Core Tip: Congenital complete heart block (CCHB) is a very rare disorder that is largely diagnosed at the fetal or infant stage. Therefore, it is infrequently diagnosed in adulthood, and the natural history of CCHB in adults is less well known. Despite the controversial literature, permanent pacing is widely recommended for the prevention of sudden death among patients with CCHB. This case illustrated an unexpectedly good course in an adult with CCHB at the onset of syncope who refused permanent pacing but led a normally active life. This suggests that CCHB in adulthood may have good outcomes and does not always require permanent pacing.