Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report
Abstract Background Eosinophilic granulomatosis with polyangiitis (EGPA) is a form of anti-neutrophil cytoplasmic antibody (ANCA) associated vasculitis characterized by eosinophil-rich granulomatous inflammation and small-to-medium vessel vasculitis associated with asthma, rhinosinusitis, and eosino...
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| Format: | Article |
| Language: | English |
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BMC
2023-04-01
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| Series: | BMC Pulmonary Medicine |
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| Online Access: | https://doi.org/10.1186/s12890-023-02415-6 |
| _version_ | 1797841213262397440 |
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| author | Isao Suzaki Akihiko Tanaka Ryo Yanai Yuki Maruyama Sawa Kamimura Kojiro Hirano Hitome Kobayashi |
| author_facet | Isao Suzaki Akihiko Tanaka Ryo Yanai Yuki Maruyama Sawa Kamimura Kojiro Hirano Hitome Kobayashi |
| author_sort | Isao Suzaki |
| collection | DOAJ |
| description | Abstract Background Eosinophilic granulomatosis with polyangiitis (EGPA) is a form of anti-neutrophil cytoplasmic antibody (ANCA) associated vasculitis characterized by eosinophil-rich granulomatous inflammation and small-to-medium vessel vasculitis associated with asthma, rhinosinusitis, and eosinophilia. EGPA is often difficult to distinguish from severe asthma and eosinophilic chronic rhinosinusitis (ECRS) in cases when there are no findings that suggest vasculitis. Dupilumab, an anti-IL-4Rα monoclonal antibody, is expected to be effective in eosinophilic airway inflammatory diseases, such as refractory asthma and chronic rhinosinusitis (CRS). Although transient eosinophilia and eosinophilic pneumoniae have been reported in patients with refractory asthma and CRS associated with dupilumab, few studies have examined the development of EGPA. Case presentation We report a case of a 61-year-old woman treated with dupilumab for refractory ECRS and eosinophilic otitis media (EOM) complicated by severe asthma. Although she had a previous history of eosinophilic pneumoniae and myeloperoxidase (MPO) ANCA positivity, there were no apparent findings of vasculitis before the initiation of dupilumab. After the second administration of dupilumab, several adverse events developed, including worsening of ECRS, EOM and asthma, and neuropathy. A blood test showed an eosoinophilia and re-elevation of MPO-ANCA levels after the administration of dupilumab. Therefore, dupilumab was discontinued owing to the development of EGPA, and prednisolone and azathioprine administration was initiated for a remission induction therapy. Conclusion To the best of our knowledge, this is the first case report that suggests that dupilumab may directly trigger the manifestation of vasculitis in patients who were previously MPO-ANCA-positive. Although the precise mechanism of how dupilumab could trigger the development of EGPA requires further elucidation, measuring MPO-ANCA in patients with multiple eosinophilic disorders before the initiation of dupilumab might be helpful when considering the possibility of a latent EGPA. When administering dupilumab to patients with a previous history of MPO-ANCA positivity, clinicians must carefully monitor and collaborate with other specialists in the pertinent fields of study for appropriate usage. |
| first_indexed | 2024-04-09T16:27:29Z |
| format | Article |
| id | doaj.art-6eb60817a19d453c8295a03cb84245f4 |
| institution | Directory Open Access Journal |
| issn | 1471-2466 |
| language | English |
| last_indexed | 2024-04-09T16:27:29Z |
| publishDate | 2023-04-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Pulmonary Medicine |
| spelling | doaj.art-6eb60817a19d453c8295a03cb84245f42023-04-23T11:08:03ZengBMCBMC Pulmonary Medicine1471-24662023-04-012311910.1186/s12890-023-02415-6Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case reportIsao Suzaki0Akihiko Tanaka1Ryo Yanai2Yuki Maruyama3Sawa Kamimura4Kojiro Hirano5Hitome Kobayashi6Department of Otorhinolaryngology, Head and Neck Surgery, School of Medicine, Showa UniversityDepartment of Medicine, Division of Respiratory Medicine and Allergology, School of Medicine, Showa UniversityDepartment of Medicine, Division of Rheumatology, School of Medicine, Showa UniversityDepartment of Otorhinolaryngology, Head and Neck Surgery, School of Medicine, Showa UniversityDepartment of Otorhinolaryngology, Head and Neck Surgery, School of Medicine, Showa UniversityDepartment of Otorhinolaryngology, Head and Neck Surgery, School of Medicine, Showa UniversityDepartment of Otorhinolaryngology, Head and Neck Surgery, School of Medicine, Showa UniversityAbstract Background Eosinophilic granulomatosis with polyangiitis (EGPA) is a form of anti-neutrophil cytoplasmic antibody (ANCA) associated vasculitis characterized by eosinophil-rich granulomatous inflammation and small-to-medium vessel vasculitis associated with asthma, rhinosinusitis, and eosinophilia. EGPA is often difficult to distinguish from severe asthma and eosinophilic chronic rhinosinusitis (ECRS) in cases when there are no findings that suggest vasculitis. Dupilumab, an anti-IL-4Rα monoclonal antibody, is expected to be effective in eosinophilic airway inflammatory diseases, such as refractory asthma and chronic rhinosinusitis (CRS). Although transient eosinophilia and eosinophilic pneumoniae have been reported in patients with refractory asthma and CRS associated with dupilumab, few studies have examined the development of EGPA. Case presentation We report a case of a 61-year-old woman treated with dupilumab for refractory ECRS and eosinophilic otitis media (EOM) complicated by severe asthma. Although she had a previous history of eosinophilic pneumoniae and myeloperoxidase (MPO) ANCA positivity, there were no apparent findings of vasculitis before the initiation of dupilumab. After the second administration of dupilumab, several adverse events developed, including worsening of ECRS, EOM and asthma, and neuropathy. A blood test showed an eosoinophilia and re-elevation of MPO-ANCA levels after the administration of dupilumab. Therefore, dupilumab was discontinued owing to the development of EGPA, and prednisolone and azathioprine administration was initiated for a remission induction therapy. Conclusion To the best of our knowledge, this is the first case report that suggests that dupilumab may directly trigger the manifestation of vasculitis in patients who were previously MPO-ANCA-positive. Although the precise mechanism of how dupilumab could trigger the development of EGPA requires further elucidation, measuring MPO-ANCA in patients with multiple eosinophilic disorders before the initiation of dupilumab might be helpful when considering the possibility of a latent EGPA. When administering dupilumab to patients with a previous history of MPO-ANCA positivity, clinicians must carefully monitor and collaborate with other specialists in the pertinent fields of study for appropriate usage.https://doi.org/10.1186/s12890-023-02415-6AsthmaDupilumabEosinophilic granulomatosis with polyangiitisEosinophilic chronic rhinosinusitis |
| spellingShingle | Isao Suzaki Akihiko Tanaka Ryo Yanai Yuki Maruyama Sawa Kamimura Kojiro Hirano Hitome Kobayashi Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report BMC Pulmonary Medicine Asthma Dupilumab Eosinophilic granulomatosis with polyangiitis Eosinophilic chronic rhinosinusitis |
| title | Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report |
| title_full | Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report |
| title_fullStr | Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report |
| title_full_unstemmed | Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report |
| title_short | Eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma: a case report |
| title_sort | eosinophilic granulomatosis with polyangiitis developed after dupilumab administration in patients with eosinophilic chronic rhinosinusitis and asthma a case report |
| topic | Asthma Dupilumab Eosinophilic granulomatosis with polyangiitis Eosinophilic chronic rhinosinusitis |
| url | https://doi.org/10.1186/s12890-023-02415-6 |
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