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<p>Immune-related adverse events (irAE) are a concern during immune checkpoint inhibitor therapy (ICI), and vasculitis is a rheumatic irAE. Granulomatosis with polyangiitis (GPA) is an anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis characterized by necrotizing granulomatosis. GPA in the mammary glands is rare and causes granulomatous mastitis. We describe an older woman with metastatic hepatocellular carcinoma who was receiving ICI therapy. She developed a rapidly growing painful mass in her right breast. Biopsy of the breast revealed necrotizing granulomatous mastitis, which implied an association with autoimmune disease. We detected c-ANCA (proteinase 3)-positivity and a nodule forming a cavity in her right lung; lung biopsy revealed granulomatosis. We diagnosed GPA, possibly arising as an irAE. Prompt initiation of prednisolone therapy led to complete resolution of clinical symptoms within 2&#xa0;weeks. This case represents the first known report of rare autoimmune mastitis with GPA flared after ICI therapy.</p>

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A rare case of mastitis involving granulomatosis with polyangiitis after immune checkpoint inhibitor therapy

  • 期刊论文

  • 发表日期：

    2025年10月20日

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  • Chieko Mishima,
  • Akane Watanabe,
  • Yuichi Motoyama,
  • Takahiro Kodama,
  • Ryu Tokui,
  • Yasufumi Sato,
  • Nanae Masunaga,
  • Masami Tsukabe,
  • Yoshiaki Sota,
  • Tetsuhiro Yoshinami,
  • Tomonori Tanei,
  • Kenzo Shimazu

摘要

Immune-related adverse events (irAE) are a concern during immune checkpoint inhibitor therapy (ICI), and vasculitis is a rheumatic irAE. Granulomatosis with polyangiitis (GPA) is an anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis characterized by necrotizing granulomatosis. GPA in the mammary glands is rare and causes granulomatous mastitis. We describe an older woman with metastatic hepatocellular carcinoma who was receiving ICI therapy. She developed a rapidly growing painful mass in her right breast. Biopsy of the breast revealed necrotizing granulomatous mastitis, which implied an association with autoimmune disease. We detected c-ANCA (proteinase 3)-positivity and a nodule forming a cavity in her right lung; lung biopsy revealed granulomatosis. We diagnosed GPA, possibly arising as an irAE. Prompt initiation of prednisolone therapy led to complete resolution of clinical symptoms within 2 weeks. This case represents the first known report of rare autoimmune mastitis with GPA flared after ICI therapy.