Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 16, 2021; 9(23): 6922-6928
Published online Aug 16, 2021. doi: 10.12998/wjcc.v9.i23.6922
Complete withdrawal of glucocorticoids after dupilumab therapy in allergic bronchopulmonary aspergillosis: A case report
Tadashi Nishimura, Tomohito Okano, Masahiro Naito, Chikashi Tsuji, Soichi Iwanaka, Yasumasa Sakakura, Taro Yasuma, Hajime Fujimoto, Corina N D'Alessandro-Gabazza, Yasuhiro Oomoto, Tetsu Kobayashi, Esteban C Gabazza, Hidenori Ibata
Tadashi Nishimura, Masahiro Naito, Chikashi Tsuji, Soichi Iwanaka, Yasumasa Sakakura, Yasuhiro Oomoto, Hidenori Ibata, Department of Pulmonary Medicine, Mie Chuo Medical Center, Tsu 514-1101, Mie, Japan
Tomohito Okano, Hajime Fujimoto, Department of Pulmonary and Critical Care Medicine, Mie University, Tsu 514-8507, Mie, Japan
Taro Yasuma, Corina N D'Alessandro-Gabazza, Esteban C Gabazza, Department of Immunology, Mie University School of Medicine, Mie University, Tsu 514-8507, Mie, Japan
Tetsu Kobayashi, Department of Pulmonary and Critical Care Medicine, Graduate School of Medicine Mie, Mie University, Tsu 514-8507, Mie, Japan
Author contributions: Nishimura T, Okano T, Tsuji C, Iwanaka S, and Sakakura Y were responsible for clinical treatment and clinical follow-up and manuscript preparation; Naito M, Ibata H, and Oomoto Y contributed with resources and acquisition of data; Fujimoto H, D'Alessandro-Gabazza CN, Yasuma T, Kobayashi T, and Gabazza EC contributed to interpreting the data and made an intellectual contribution to the manuscript's preparation. All Authors reviewed and approved the final manuscript.
Informed consent statement: Informed consent was obtained from the subject involved in the study.
Conflict-of-interest statement: None of the authors declared no conflict of interest concerning this case report.
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: Esteban C Gabazza, MD, PhD, Director, Full Professor, Department of Immunology, Mie University School of Medicine, Mie University, Edobashi 2-174, Tsu 514-8507, Japan. gabazza@doc.medic.mie-u.ac.jp
Received: April 25, 2021
Peer-review started: April 25, 2021
First decision: June 6, 2021
Revised: June 8, 2021
Accepted: June 22, 2021
Article in press: June 22, 2021
Published online: August 16, 2021
Processing time: 102 Days and 6.5 Hours
Abstract
BACKGROUND

Allergic bronchopulmonary aspergillosis (ABPA) is an allergic reaction to Aspergillus species that aggravates bronchial asthma. Previous studies demonstrated the glucocorticoid-sparing effect of dupilumab in patients with ABPA. There is no report of complete withdrawal of glucocorticoids after dupilumab.

CASE SUMMARY

The patient was a 54-year-old woman with bronchial asthma treated with inhaled corticosteroids and a long-acting beta-2 agonist. She consulted our institution for productive cough and fever in March 2017. Chest computed tomography scan revealed mucoid impaction, and the bronchial lavage fluid culture was positive for Aspergillus fumigatus. The diagnosis was ABPA. The patient was treated with oral glucocorticoids from April 2017 to November 2017. In January 2019, she had bronchial asthma exacerbation, and a chest computed tomography scan showed recurrent mucoid impaction. She was treated with oral glucocorticoids and itraconazole. In February 2020, during tapering of oral glucocorticoid, she had the third episode of bronchial asthma exacerbation and a mucoid impaction. The patient was treated with dupilumab in addition to oral glucocorticoid and itraconazole. The clinical response improved, and oral glucocorticoid was discontinued in June 2020.

CONCLUSION

This is the first case of ABPA in which complete withdrawal of glucocorticoid was possible after treatment with dupilumab.

Keywords: Allergic bronchopulmonary aspergillosis; Dupilumab; Asthma; Interleukin-13; Interleukin-4; Case report

Core Tip: Allergic bronchopulmonary aspergillosis (ABPA) is an allergic reaction to antigen from Aspergillus species that causes exacerbation of bronchial asthma, eosinophilic pneumonia, and bronchiectasis. Conventional therapy includes the administration of oral glucocorticoids and other antiasthmatic therapy. However, there are intractable cases showing repeated clinical exacerbations of bronchial asthma despite therapy with oral glucocorticoids. Previous reports have shown that the addition of monoclonal antibodies against interleukin (IL)-4/IL-13 or dupilumab to the treatment is effective and allows the reduction in the dose of glucocorticoids in ABPA patients. However, there is no report of complete withdrawal of oral glucocorticoids after therapy with dupilumab in these patients. Here, we report the first case of ABPA in which complete withdrawal of oral glucocorticoid was possible after treatment with dupilumab.