Matsuhisa Y, Kenzaka T, Shimizu H, Hirose H, Gotoh T. Recurrence of unilateral angioedema of the tongue: A case report. World J Clin Cases 2023; 11(27): 6603-6612 [PMID: 37900225 DOI: 10.12998/wjcc.v11.i27.6603]
Corresponding Author of This Article
Yuki Matsuhisa, MD, Attending Doctor, Chief Doctor, Department of General Medicine, Center for Community Medicine in North-Western Gifu Prefecture National Health Insurance Shirotori Hospital, No. 1205-1 Tamezani, Shirotori-cho, Gujo 501-5122, Japan. m02084ym@jichi.ac.jp
Research Domain of This Article
Allergy
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: http://creativecommons.org/licenses/by-nc/4.0/
Yuki Matsuhisa, Hironori Shimizu, Hideo Hirose, Tadao Gotoh, Department of General Medicine, Center for Community Medicine in North-Western Gifu Prefecture National Health Insurance Shirotori Hospital, Gujo 501-5122, Japan
Yuki Matsuhisa, Department of Pediatrics, Center for Community Medicine in North-Western Gifu Prefecture National Health Insurance Shirotori Hospital, Gujo 501-5122, Japan
Tsuneaki Kenzaka, Department of Internal Medicine, Hyogo Prefectural Tamba Medical Center, Tamba 669-3495, Japan
Tsuneaki Kenzaka, Division of Community Medicine and Career Development, Kobe University Graduate School of Medicine, Kobe 652-0032, Japan
Author contributions: Shimizu H, Hirose H, and Gotoh T contributed to data analysis; Matsuhisa Y contributed to investigation; Matsuhisa Y and Kenzaka T contributed to writing-original draft preparation; Matsuhisa Y, Kenzaka T; Shimizu H, Hirose H, and Gotoh T contributed to writing-review and editing; All authors have read and agreed to the published version of the manuscript.
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 conflict 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: Yuki Matsuhisa, MD, Attending Doctor, Chief Doctor, Department of General Medicine, Center for Community Medicine in North-Western Gifu Prefecture National Health Insurance Shirotori Hospital, No. 1205-1 Tamezani, Shirotori-cho, Gujo 501-5122, Japan. m02084ym@jichi.ac.jp
Received: July 2, 2023 Peer-review started: July 2, 2023 First decision: August 16, 2023 Revised: August 24, 2023 Accepted: August 31, 2023 Article in press: August 31, 2023 Published online: September 26, 2023
Abstract
BACKGROUND
Angioedema is a disorder characterized by edema of the face, lips, tongue, and extremities due to increased vascular permeability. Angioedema of the tongue usually occurs bilaterally, and the incidence of unilateral angioedema of the tongue is rare. This study reports a rare case of unilateral angioedema of the tongue with no identifiable cause and repeated recurrence even after discontinuation of an angiotensin-converting enzyme inhibitor.
CASE SUMMARY
The patient was a 65-year-old woman with pre-existing hypertension and hyperlipidemia and had been receiving 20 mg/d of lisinopril. She was diagnosed with angioedema due to unilateral swelling of the tongue. No airway obstruction was observed, and the symptoms improved following the administration of 0.3 mg of epinephrine intramuscularly and 125 mg of methylprednisolone intravenously. Although lisinopril was discontinued, unilateral angioedema of the tongue continued to recur every 2-5 mo, with the symptoms improving following the administration of prednisolone and an antihistamine. Daily oral administration of 500 mg of tranexamic acid after dinner prevented the recurrence of angioedema.
CONCLUSION
Careful monitoring and identification of the underlying mechanism play a crucial role in the treatment of angioedema.
Core Tip: This study describes the case of a 65-year-old woman who presented with unilateral angioedema of the tongue with no identifiable cause and who showed repeated recurrence of angioedema after discontinuation of an angiotensin-converting enzyme (ACE) inhibitor. She had a history of hypertension and hyperlipidemia for 32 years and had been receiving 20 mg/d of lisinopril for 32 years. The ACE inhibitor was suspected as the cause of angioedema; therefore, its use was discontinued. However, angioedema of the left unilateral tongue continued to recur. Recurrence of the unilateral angioedema did not occur following the administration of 500 mg of tranexamic acid.
