Case Report
Copyright ©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 16, 2025; 13(11): 98128
Published online Apr 16, 2025. doi: 10.12998/wjcc.v13.i11.98128
Rapid improvement in postpartum pulmonary hypertension associated with hereditary hemorrhagic telangiectasia: A case report and review of literature
Sheng-Yu Hao, Yaxiaerjiang Muhetaer, Xin Zheng, Yu-Liang Long, Jie-Qiong Song, Ming Zhong
Sheng-Yu Hao, Yaxiaerjiang Muhetaer, Xin Zheng, Jie-Qiong Song, Ming Zhong, Department of Critical Care Medicine, Zhongshan Hospital Affiliated to Fudan University, Shanghai 200032, China
Yu-Liang Long, Department of Cardiology, Zhongshan Hospital Affiliated to Fudan University, Shanghai 200032, China
Co-first authors: Sheng-Yu Hao and Yaxiaerjiang Muhetaer.
Co-corresponding authors: Jie-Qiong Song and Ming Zhong.
Author contributions: Hao SY, Muhetaer Y, and Zheng X collected the patient’s information, summarized the literature data, and wrote the manuscript; Hao SY and Muhetaer Y contributed equally to this article, they are the co-first authors of this manuscript; Long YL followed up with the patient and collected the patient’s information; Song JQ and Zhong M were the major contributors in revising the manuscript, they contributed equally to this article, they are the co-corresponding authors of this manuscript; and all authors have read and approved the final manuscript.
Supported by the Shanghai Sailing Program, No. 21YF1440300 and No. 22YF1407700; and the National Natural Science Foundation of China, No. 82200061.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
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: Ming Zhong, PhD, Department of Critical Care Medicine, Zhongshan Hospital Affiliated to Fudan University, No. 180 Fenglin Road, Shanghai 200032, China. zhongming2022@163.com
Received: June 18, 2024
Revised: November 18, 2024
Accepted: December 10, 2024
Published online: April 16, 2025
Processing time: 190 Days and 18.4 Hours
Core Tip

Core Tip: This case report examines pulmonary vasodilators in a postpartum patient with hereditary hemorrhagic telangiectasia and pulmonary arterial hypertension carrying an activin receptor-like kinase 1 mutation. Despite treatments with diuretics, sildenafil, and nitric oxide, the efficacy was limited. Inhaled iloprost, however, rapidly relieved symptoms and improved hemodynamics. The study underscores the importance of genotype in treatment responses and highlights the need for personalized management of hereditary hemorrhagic telangiectasia-related pulmonary arterial hypertension, especially during pregnancy and postpartum. Further research is needed to explore underlying mechanisms and drug interactions.