Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Nov 16, 2022; 10(32): 11898-11907
Published online Nov 16, 2022. doi: 10.12998/wjcc.v10.i32.11898
Successful treatment of pulmonary hypertension in a neonate with bronchopulmonary dysplasia: A case report and literature review
Jiao Li, Jing Zhao, Xiao-Yan Yang, Jing Shi, Hai-Ting Liu
Jiao Li, Jing Zhao, Xiao-Yan Yang, Hai-Ting Liu, Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Jing Shi, Neonatal Ward, West China Second University Hospital, Sichuan University, Chengdu 610066, Sichuan Province, China
Author contributions: Li J contributed to the design and drafting of the manuscript and the collection of clinical data; Zhao J and Yang XY contributed to the clinical data analysis; Liu HT and Shi J supervised and interpreted the data; All authors have read and approved the final manuscript.
Supported by The Clinical Research Fund of West China Second University Hospital, Sichuan University, No. KL109 (to Li J), No. KL014 (to Yang XY), and No. KL075 (to Shi J).
Informed consent statement: Written informed consent was obtained from the patient’s parents prior to the study.
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: Hai-Ting Liu, PhD, Attending Doctor, Department of Pediatrics, West China Second University Hospital, Sichuan University, No. 20 Section 3, South Renmin Road, Chengdu 610000, Sichuan province, China. liuhaiting9@163.com
Received: June 17, 2022
Peer-review started: June 17, 2022
First decision: August 21, 2022
Revised: September 3, 2022
Accepted: October 11, 2022
Article in press: October 11, 2022
Published online: November 16, 2022
Abstract
BACKGROUND

Pulmonary hypertension (PH) is a severe complication of bronchopulmonary dysplasia (BPD) in premature neonates and is closely related to prognosis. However, there is no effective and safe treatment for PH due to BPD in infants. Successful treatment for cases of BPD-associated PH with Tadalafil combined with bosentan is rare. This case may make a significant contribution to the literature because PH is difficult to manage as a serious complication of BPD in preterm infants. Mortality is high, especially when it is complicated by heart failure.

CASE SUMMARY

An extremely premature neonate with a gestational age of 26+5 wk and birth weight of 0.83 kg was diagnosed with BPD associated with PH; oral sildenafil did not improve the PH. The infant experienced sudden cardiac arrest and serious heart failure with severe PH. After a series of treatments, including cardiopulmonary resuscitation, mechanical ventilation, and inhaled nitric oxide (iNO), the respiratory and circulatory status improved but the pulmonary artery pressure remained high. Then oral sildenafil was replaced with oral tadalafil and bosentan; pulmonary artery pressure improved, and the infant recovered at our hospital. After 2 years of follow-up, she is in good condition, without any cardiovascular complications.

CONCLUSION

INO can effectively improve the respiratory and circulatory status of infants with PH associated with premature BPD. B-type natriuretic peptide should be routinely measured during hospitalization to evaluate the risk and prognosis of BPD-associated PH in preterm infants. Tadalafil combined with bosentan for the treatment of PH associated with premature BPD was better than sildenafil in this case. Further studies are needed to explore the efficacy and safety of different vasodilators in the treatment of PH associated with premature BPD.

Keywords: Premature, Bronchopulmonary dysplasia, Pulmonary hypertension, Treatment, Case report

Core Tip: Pulmonary hypertension (PH) is difficult to manage as a serious complication of bronchopulmonary dysplasia (BPD) in preterm infants. Mortality is high, especially when it is complicated by heart failure. Valuable and noninvasive biomarkers, such as B-type natriuretic peptide, should be routinely measured during hospitalization to evaluate the risk and prognosis of BPD-associated PH in preterm infants. Inhaled nitric oxide can effectively improve the respiratory and circulatory status of infants with PH associated with premature BPD. Tadalafil combined with bosentan for the treatment of PH associated with premature BPD was better than sildenafil in this case.