Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 16, 2023; 11(26): 6268-6273
Published online Sep 16, 2023. doi: 10.12998/wjcc.v11.i26.6268
Recurrent hemoptysis in pediatric bronchial Dieulafoy’s disease with inferior phrenic artery supply: A case report
Fang Wang, Jiao Tang, Mou Peng, Pu-Jue Huang, Li-Juan Zhao, Yin-Yue Zhang, Tao Wang
Fang Wang, Jiao Tang, Mou Peng, Pu-Jue Huang, Li-Juan Zhao, Yin-Yue Zhang, Tao Wang, Department of Pediatric Cardiology, West China Second University Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Jiao Tang, Pu-Jue Huang, Department of Pediatrics, The First People's Hospital of Longquanyi District, Chengdu 610041, Sichuan Province, China
Li-Juan Zhao, Department of Pediatrics, Western Theater General Hospital, Chengdu 610041, Sichuan Province, China
Tao Wang, Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Chengdu 610041, Sichuan Province, China
Tao Wang, Key Laboratory of Development and Diseases of Women and Children of Sichuan Province, Chengdu 610041, Sichuan Province, China
Author contributions: Wang F, Tang J, and Peng M contributed equally to this work; Wang F and Tang J analyzed the case and wrote the manuscript; Peng M contributed new reagents and analytic tools, and carried out the definition of intellectual content; Huang PJ and Zhao LJ were involved in study concept, study design, and manuscript preparation; Wang T performed the role of guarantor for the integrity of the entire study and was involved in the literature research and manuscript editing; and all authors have read and approved the final manuscript.
Supported by the National Natural Science Foundation of China, No. 81701888; and Science-Technology Support Plan Projects of Sichuan Province, No. 2019YFS0239 and No. 2023YFS0206.
Informed consent statement: Informed written consent was obtained from the patient for the publication of this report and any accompanying images.
Conflict-of-interest statement: All the authors declare that they have no conflict of interest to disclose.
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: Tao Wang, MD, Chief Physician, Department of Pediatric Cardiology, West China Second University Hospital, Sichuan University, No. 20, 3rd Section, South Renmin Road, Chengdu 610041, Sichuan Province, China. 44871875@qq.com
Received: June 9, 2023
Peer-review started: June 9, 2023
First decision: July 28, 2023
Revised: August 9, 2023
Accepted: August 23, 2023
Article in press: August 23, 2023
Published online: September 16, 2023
Processing time: 91 Days and 2.6 Hours
Abstract
BACKGROUND

Bronchial Dieulafoy’s disease (BDD) is characterized by the erosion of an anomalous artery in the submucosa of the bronchus. The etiology of pediatric BDD is mainly congenital dysplasia of bronchus and pulmonary arteries, which is different from chronic inflammatory injury of the airway in adult patients. The internal thoracic artery, subclavian artery, and intercostal artery are known to be involved in the blood supply to the BDD lesion in children.

CASE SUMMARY

We report a case of BDD in a 4-year-old boy with recurrent hemoptysis for one year. Selective angiography showed a dilated right bronchial artery, and anastomosis of its branches with the right lower pulmonary vascular network. Bronchoscopy showed nodular protrusion of the bronchial mucosa with a local scar. Selective embolization of the bronchial artery was performed to stop bleeding. One month after the first intervention, the symptoms of hemoptysis recurred. A computed tomography angiogram (CTA) showed another tortuous and dilated feeding artery in the right lower lung, which was an abnormal ascending branch of the inferior phrenic artery (IPA). The results of angiography were consistent with the CTA findings. The IPA was found to be another main supplying artery, which was not considered during the first intervention. Finally, the IPA was also treated by microsphere embolization combined with coil interventional closure. During the one-year follow-up, the patient never experienced hemoptysis.

CONCLUSION

The supplying arteries of the bleeding lesion in children with BDD may originate from multiple different aortopulmonary collateral arteries, and the IPA should be considered to reduce missed diagnosis. CTA is a noninvasive radiological examination for the screening of suspected vessels, which shows a high coincidence with angiography, and can serve as the first choice for the diagnosis of BDD.

Keywords: Hemoptysis; Child; Bronchial Dieulafoy’s disease; Inferior phrenic artery; Interventional treatment; Case report

Core Tip: Bronchial Dieulafoy’s disease (BDD) in children is mostly caused by congenital dysplasia of bronchus and/or pulmonary arteries, which is different from chronic inflammatory injury of the airway in adult patients. We report a 4-year-old male BDD patient with repeated hemoptysis. Selective embolization of the responsible bronchial artery was performed to stop bleeding in the first time. One month after the interventional operation, however, the patient repeated the symptoms of hemoptysis. And the inferior phrenic artery (IPA) was found to be another supplying artery that caused hemoptysis in addition to the bronchial arteries. Finally, the artery was also treated by microsphere embolization with coil interventional closure. The responsible arteries of the bleeding lesion in children with BDD may originate from multiple different aortopulmonary collateral arteries, and the IPA should be considered to reduce missed diagnosis. Computed tomography angiogram is a noninvasive radiological examination for the screening of the responsible vessels, which shows a high coincidence with angiography, and can serve as the first choice for the diagnosis of BDD in suspected patients.