Izhakian S, Korlansky M, Rosengarten D, Bruckheimer E, Kramer MR. Pulmonary artery stent thrombosis and symptomatic pulmonary hypertension following COVID-19 infection in Alagille patient: A case report. World J Clin Cases 2025; 13(9): 96897 [DOI: 10.12998/wjcc.v13.i9.96897]
Corresponding Author of This Article
Shimon Izhakian, MD, PhD, Doctor, Senior Lecturer, Pulmonary Institute, Rabin Medical Center, 39 Jabotinski St., Petach Tikva 49100, Israel. shimixyz@gmail.com
Research Domain of This Article
Respiratory System
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/
Shimon Izhakian, Miriam Korlansky, Dror Rosengarten, Mordechai Reuven Kramer, Pulmonary Institute, Rabin Medical Center, Petach Tikva 49100, Israel
Elchanan Bruckheimer, Cardiac Catheterization Laboratory, Schneider Children's Medical Center, Petach Tikva 49100, Israel
Co-first authors: Shimon Izhakian and Miriam Korlansky.
Author contributions: Izhakian S and Korlansky M contributed equally to the acquisition and interpretation of the data; Izhakian S and Korlansky M contributed equally to the critical revision of the manuscript for important intellectual content; Izhakian S, Korlansky M, Rosengarten D, Bruckheimer E and Kramer MR contributed to the drafting of the manuscript and approved the final version.
Informed consent statement: Written informed consent was obtained from the patient for publication of this manuscript and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest related to this work.
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: Shimon Izhakian, MD, PhD, Doctor, Senior Lecturer, Pulmonary Institute, Rabin Medical Center, 39 Jabotinski St., Petach Tikva 49100, Israel. shimixyz@gmail.com
Received: May 17, 2024 Revised: October 8, 2024 Accepted: December 2, 2024 Published online: March 26, 2025 Processing time: 208 Days and 20.6 Hours
Abstract
BACKGROUND
Alagille syndrome is a multisystem disease that results in various vascular anomalies, commonly involving the cardiac and pulmonary systems. To the best of our knowledge, there is no literature regarding the cardiovascular outcomes of these patients in association with coronavirus disease 2019 (COVID-19).
CASE SUMMARY
A 34-year-old woman with a history of Alagille syndrome who underwent successful atrial septal defect with partial anomalous pulmonary veins and patent ductus arteriosus repair, as well as left pulmonary artery catheterization and stenting in childhood due to pulmonary stenosis. The patient was without any respiratory symptoms and was a dancer prior to contracting COVID-19. Several weeks after her COVID-19 infection, she developed left pulmonary artery stent thrombosis and subsequent symptomatic pulmonary hypertension. A treatment strategy of anticoagulation alongside pharmacological agents for pulmonary hypertension for 3 months followed by balloon pulmonary artery angioplasty to reopen the stenosis was unsuccessful.
CONCLUSION
In the era of COVID-19, patients with pulmonary vascular malformations and endovascular stents are at an increased risk for chronic thromboembolic disease. Patients may benefit from prophylactic antiplatelet or anticoagulation therapy. Stent thrombosis is a devastating phenomenon and should be treated urgently and aggressively with balloon pulmonary angioplasty, and/or a thrombolytic agent.
Core Tip: We suggest that in this era of coronavirus disease 2019, patients with endovascular stents and known pulmonary vessel malformations be considered for preventive anticoagulation or antiplatelet therapy, since a thrombotic event in such patients may result in secondary pulmonary hypertension and devastating, irreversible damage. Furthermore, patients with thrombosed stents should be treated aggressively and quickly once a clot is diagnosed.
