Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 6, 2021; 9(22): 6435-6442
Published online Aug 6, 2021. doi: 10.12998/wjcc.v9.i22.6435
Manifestation of severe pneumonia in anti-PL-7 antisynthetase syndrome and B cell lymphoma: A case report
Xuan-Li Xu, Ru-Hui Zhang, Yue-Hong Wang, Jian-Ying Zhou
Xuan-Li Xu, Ru-Hui Zhang, Yue-Hong Wang, Jian-Ying Zhou, Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310003, Zhejiang Province, China
Author contributions: Xu XL and Zhang RH were the patient’s physician, reviewed the literature and contributed to manuscript drafting; Wang YH and Zhou JY were responsible for the revision of the manuscript for important intellectual content; all authors issued final approval for the version to be submitted.
Supported by National Natural Science Foundation of China, No. 81900020; and Natural Science Foundation of Zhejiang Province, China, No. LQ19H160020.
Informed consent statement: Study participant and his legal guardian, provided informed written consent prior to study enrollment.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Jian-Ying Zhou, MD, Chief Doctor, Professor, Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital, Zhejiang University School of Medicine, No. 9 Qingchun Road, Hangzhou 310003, Zhejiang Province, China. zjyhz@zju.edu.cn
Received: April 3, 2021
Peer-review started: April 3, 2021
First decision: April 28, 2021
Revised: May 8, 2021
Accepted: May 24, 2021
Article in press: May 24, 2021
Published online: August 6, 2021
Processing time: 115 Days and 13.3 Hours
Abstract
BACKGROUND

Antisynthetase syndrome (ASS) is characterized by the presence of antisynthetase antibodies coupled with clinical findings such as fever, polymyositis-dermatomyositis and interstitial lung disease. It is, however, rare to observe ASS association with B cell lymphoma presenting severe pneumonia as the first clinical manifestation.

CASE SUMMARY

We evaluated a 59-year-old male patient who presented with cough with sputum, shortness of breath and fever for 13 d. A chest computed tomography radiograph revealed bilateral diffuse ground-glass infiltrates in both upper fields, left lingual lobe and right middle lobe. Initially, the patient was diagnosed with severe community-acquired pneumonia and respiratory failure. He was empirically treated with broad-spectrum antibiotics, without improvement. Further analysis showed an ASS panel with anti-PL7 antibodies. Besides, electromyography evaluation demonstrated a manifestation of myogenic damage, while deltoid muscle biopsy showed irregular muscle fiber bundles especially abnormal lymphocyte infiltration. In addition, bone marrow biopsy revealed high invasive B cell lymphoma. Thus, the patient was diagnosed with a relatively rare anti–PL7 antibody positive ASS associated with B cell lymphoma.

CONCLUSION

This case highlights that rapidly progressive lung lesions and acute hypoxemic respiratory failure associated with heliotrope rash and extremely high lactate dehydrogenase level should be considered as the characteristics of non-infectious diseases, especially ASS and B cell lymphoma.

Keywords: Antisynthetase syndrome; PL7; B cell lymphoma; Severe pneumonia; Case report

Core Tip: Antisynthetase syndrome (ASS) is a unique subset of inflammatory myopathy. Patients with inflammatory myopathies carry a higher risk of developing neoplasms, most commonly adenocarcinoma but not lymphoma or other hematologic neoplasms. However, data on the association between ASS and malignancy remain very scant. We present here a rare case of severe pneumonia and acute hypoxemic respiratory failure as the first indicator for anti-PL-7 ASS accompanied by B cell lymphoma.