Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 16, 2022; 10(35): 13074-13080
Published online Dec 16, 2022. doi: 10.12998/wjcc.v10.i35.13074
Pulmonary sarcoidosis: A novel sequelae of drug reaction with eosinophilia and systemic symptoms: A case report
Yu-Qi Hu, Chen-Yang Lv, Ai Cui
Yu-Qi Hu, Chen-Yang Lv, Ai Cui, Department of Pulmonary and Critical Care Medicine, Beijing Chao-Yang Hospital, Capital Medical University, Beijing 100020, China
Chen-Yang Lv, Department of Pulmonary and Critical Care Medicine, The First Hospital of Fangshan District, Beijing 102499, China
Author contributions: Hu YQ and Lv CY collected the data, imaging, and wrote the initial draft of the manuscript; Cui A was the primary physician during the patient’s hospital stay and was responsible for overseeing the report and editing the manuscript; all authors read and approved the final manuscript.
Informed consent statement: All study participants, or their legal guardian, provided informed written consent prior to study enrollment.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised following 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: Ai Cui, MD, Chief Physician, Professor, Department of Pulmonary and Critical Care Medicine, Beijing Chao-Yang Hospital, Capital Medical University, No. 8 Gongren Tiyuchang Nanlu, Chaoyang District, Beijing 100020, China. cuiai@ccmu.edu.cn
Received: September 14, 2022
Peer-review started: September 14, 2022
First decision: November 4, 2022
Revised: November 13, 2022
Accepted: November 23, 2022
Article in press: November 23, 2022
Published online: December 16, 2022
Processing time: 90 Days and 20.1 Hours
Abstract
BACKGROUND

Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is an uncommon yet serious adverse drug hypersensitivity reaction with the presentations including rash, fever, lymphadenopathy, and internal organ involvement. Sarcoidosis is a systematic granulomatous disease with unknown etiology. We herein report a case of pulmonary sarcoidosis secondary to allopurinol-induced DRESS.

CASE SUMMARY

A 37-year-old man with a history of hyperuricemia was treated with allopurinol for three weeks at a total dose of 7000 milligrams before developing symptoms including anorexia, fever, erythematous rash, and elevated transaminase. The patient was diagnosed with DRESS and was treated with prednisone for 6 mo until all the symptoms completely resolved. Three months later, the patient presented again because of a progressively worsening dry cough. His chest computed tomography images showed bilateral lung parenchyma involvement with lymph node enlargement, which was confirmed to be nonnecrotizing granuloma by pathological examination. Based on radiologic and pathological findings, he was diagnosed with sarcoidosis and was restarted on treatment with prednisone, which was continued for another 6 mo. Reexamination of chest imaging revealed complete resolution of parenchymal lung lesions and a significant reduction in the size of the mediastinal and hilar lymph nodes. Following a 6-month follow-up of completion of treatment, the patient's clinical condition remained stable with no clinical evidence of relapse.

CONCLUSION

This is the first case in which pulmonary sarcoidosis developed as a late complication of allopurinol-induced DRESS. The case indicated that the autoimmune reaction of DRESS may play an important role in the pathogenesis of sarcoidosis.

Keywords: Pulmonary sarcoidosis, Drug reaction with eosinophilia and systemic symptoms, Autoimmune sequelae, Allopurinol, Case report

Core Tip: Sarcoidosis is a clinical challenge due to its less understood etiology and heterogeneous manifestations in most cases. Here, we report a unique case of pulmonary sarcoidosis that developed as a prolonged symptom of allopurinol-induced drug reaction with eosinophilia and systemic symptoms (DRESS). The case indicated that the autoimmune reaction of DRESS may play an important role in the pathogenesis of sarcoidosis.