Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 26, 2020; 8(12): 2662-2666
Published online Jun 26, 2020. doi: 10.12998/wjcc.v8.i12.2662
Diffuse alveolar hemorrhage with histopathologic manifestations of pulmonary capillaritis: Three case reports
Jun Xie, Ying-Yue Zhao, Jing Liu, Guang-Min Nong
Jun Xie, Ying-Yue Zhao, Jing Liu, Guang-Min Nong, Department of Pediatrics, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
Author contributions: Xie J, Zhao YY, and Nong GM conceptualized and designed the study; Xie J and Zhao YY drafted the initial manuscript; Liu J designed the data collection instruments, collected the data, and carried out the initial analyses; Xie J, Zhao YY, and Liu J reviewed and revised the manuscript; Nong GM coordinated and supervised the data collection, and critically reviewed the manuscript for important intellectual content; All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
Informed consent statement: Informed written consent was obtained from the patients for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors have no potential conflicts of interest to disclose.
CARE Checklist (2016) statement: 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: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Guang-Min Nong, MD, Professor, Department of Pediatrics, the First Affiliated Hospital of Guangxi Medical University, No. 6 Shuangyong Road, Nanning 530021, Guangxi Zhuang Autonomous Region, China. ngm8525@163.com
Received: February 26, 2020
Peer-review started: February 26, 2020
First decision: April 21, 2020
Revised: April 28, 2020
Accepted: May 21, 2020
Article in press: May 21, 2020
Published online: June 26, 2020
Abstract
BACKGROUND

Diffuse alveolar hemorrhage (DAH) is a multicause pulmonary capillary hemorrhage or pulmonary vascular small vessel injury (mainly capillaries, including arteries and veins), causing pulmonary microcirculation blood to accumulate in the alveolar space. DAH is classified by the histological absence or presence of pulmonary capillaritis (PC) and is rarely reported in the literature.

CASE SUMMARY

This is a report of three girls aged 6-11 years with DAH and PC. Two patients had decreased hemoglobin and one had increased erythrocyte sedimentation rate. High-resolution computed tomography showed bilateral diffuse pulmonary infiltrate, and diagnosis of PC was confirmed by lung biopsy. Immunofluorescence test in one case showed granular IgG and a small amount of granular IgA deposit on the alveolar walls, and was negative in the other two cases, describing isolated pauci-immune PC. Treatment was with glucocorticoid alone or combination with immunosuppressants, and the symptoms resolved in all patients.

CONCLUSION

PC is classified as isolated and immune-mediated PC associated with systemic disease. It can be controlled in most children with glucocorticoid alone or combined with immunosuppressants.

Keywords: Diffuse alveolar hemorrhage, Pulmonary capillaritis, Glucocorticoid, Immunosuppressant, Lung biopsy, Case report

Core tip: This is a report of three children with diffuse alveolar hemorrhage and pulmonary capillaritis who presented with recurrent hemoptysis: One was antinuclear antibody-positive and two were antibody-negative. The condition can be controlled in patients using glucocorticoid alone or combination of immunosuppressant treatment.