Xie J, Zhao YY, Liu J, Nong GM. Diffuse alveolar hemorrhage with histopathologic manifestations of pulmonary capillaritis: Three case reports. World J Clin Cases 2020; 8(12): 2662-2666 [PMID: 32607347 DOI: 10.12998/wjcc.v8.i12.2662]
Corresponding Author of This Article
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
Research Domain of This Article
Pediatrics
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/
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 Processing time: 119 Days and 0.3 Hours
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.
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.