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Case Reports
. 2022;61(18):2803-2808.
doi: 10.2169/internalmedicine.8447-21. Epub 2022 Sep 15.

Remission of Granulomatosis with Polyangiitis Only After Resection of a Pulmonary Nodule

Ryuichiro Kanda  1 , Kazuhisa Nakano  2 , Aya Nawata  3 , Shigeru Iwata  1 , Shingo Nakayamada  1 , Yoshiya Tanaka  1
Affiliations
Case Reports

Remission of Granulomatosis with Polyangiitis Only After Resection of a Pulmonary Nodule

Ryuichiro Kanda et al. Intern Med. 2022.

Abstract

Granulomatosis with polyangiitis (GPA) is characterized by necrotizing granulomatous lesions and is classified as ANCA-associated vasculitis (AAV). We herein report a case of GPA that was remitted by resection of a pulmonary lesion without immunosuppressive therapy. We detected activated neutrophils and neutrophil extracellular traps (NET) formation in resected lung tissue by immunofluorescence. Activated neutrophils and NETs might be involved in the pathophysiology of AAV and induce the vicious cycle of ANCAs and NETs. In cases of GPA with no other severe lesions, the reevaluation of the disease activity after diagnostic resection is crucial for considering the need for immunosuppressive therapy.

Keywords: ANCA-associated vasculitis (AAV); NETosis; granulomatosis with polyangiitis (GPA); neutrophil extracellular traps (NETs).

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Conflict of interest statement

Author's disclosure of potential Conflicts of Interest (COI).

Kazuhisa Nakano: Honoraria, Bristol-Myers, Sanofi, AbbVie, Eisai, Eli Lilly, Chugai, Pfizer, Takeda, and Mitsubishi-Tanabe; Research funding, Mitsubishi-Tanabe and Eisai. Shingo Nakayamada: Honoraria, Bristol-Myers, Pfizer, GlaxoSmithKline, Sanofi, Chugai, Astellas, Asahi-kasei, and Boehringer Ingelheim; Research funding, Mitsubishi-Tanabe, Novartis and MSD. Yoshiya Tanaka: Honoraria, Daiichi-Sankyo, Eli Lilly, Novartis, YL Biologics, Bristol-Myers, Eisai, Chugai, Abbvie, Astellas, Pfizer, Sanofi, Asahi-kasei, GSK, Mitsubishi-Tanabe, Gilead, Janssen, Eli Lilly, Daiichi-Sankyo, Taisho, Ayumi, Sanofi, GSK, and Abbvie; Research funding, Abbvie, Mitsubishi-Tanabe, Chugai, Asahi-Kasei, Eisai, Takeda, and Daiichi-Sankyo.

Figures

Figure 1.
Figure 1.
CT findings before and after surgery: (A-C) CT findings on October, 2019 (before surgery): There were two pulmonary nodular shadows (S1 and S6) and stenosis of the left ear canal. (D-F) CT findings on January, 2020 (after surgery): The lung nodule (S1) was resected surgically (red arrow). After that, another nodule (S6) disappeared two weeks later, and the stenosis of left ear canal improved without treatment (blue arrow and round). (G-H) CT findings on November, 2020: There was no relapse, even at one year after the surgery.
Figure 2.
Figure 2.
Histopathological findings of the resected lung tissue (S1): (Left) Severe inflammatory infiltration including neutrophils is seen in the alveolar walls [Hematoxylin and Eosin (H&E) staining, ×20]. (Middle) An epithelioid cell granuloma with multinucleated giant cells (black arrows) is detected (H&E staining, ×200). (Right) The small pulmonary artery is experiencing neutrophil infiltration (black arrows) (H&E staining, ×400).
Figure 3.
Figure 3.
Clinical course: After partial lung resection, her symptoms of a fever, cough, and ear fullness improved, and the levels of CRP and myeloperoxidase (MPO)-ANCA decreased gradually.
Figure 4.
Figure 4.
Immunofluorescence staining in the resected lung tissue (S1): DNA (blue) and histone (red) staining revealed formation of NETs that had been released by neutrophils (green) in the alveolar wall. The origin of Lung 1 and Lung 2 is the same S1 lung segment.
See this image and copyright information in PMC

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