CASE REPORT article
Front. Immunol.
Sec. Autoimmune and Autoinflammatory Disorders: Autoinflammatory Disorders
Volume 16 - 2025 | doi: 10.3389/fimmu.2025.1600250
This article is part of the Research TopicAutoinflammatory novelties: from pathogenic mechanisms to clinical and therapeutic implicationsView all 9 articles
Case Report: Dual Immunomodulatory and Hematologic Benefits of Rituximab in Refractory Anemia of ANCA-Associated Vasculitis
Provisionally accepted
- Ningbo No.2 hospital, Ningbo, Zhejiang Province, China
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Background: Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) is a systemic autoimmune disease characterized by necrotizing small-vessel inflammation, frequently complicated by severe anemia and progressive renal injury. Anemia, affecting 73-92% of AAV patients, arises from multifactorial mechanisms including renal dysfunction, chronic inflammation, and iron dysregulation. Despite conventional immunosuppressive therapies, refractory anemia remains a significant challenge, with limited strategies targeting inflammation-driven hepcidin dysregulation. Case Presentation: A 56-year-old woman presented with myeloperoxidase-specific antineutrophil cytoplasmic antibody (MPO-ANCA) positive AAV, transfusion-dependent anemia (hemoglobin: 56 g/L), and advanced chronic kidney disease with 55% tubulointerstitial atrophy. Initial management included cyclophosphamide, glucocorticoids, erythropoietin, and transfusions, yielding only a transient rise in hemoglobin (Hb) that rapidly declined despite treatment. Following the initiation of rituximab (RTX), her Hb level improved to 88 g/L within four weeks and normalized to 127 g/L after four biweekly infusions (500 mg each). Concurrently, MPO-ANCA titers decreased from 1:1280 to 1:80, and pulmonary infiltrates resolved. However, renal function remained impaired (serum creatinine: 229 µmol/L) due to irreversible fibrosis. Conclusions: This case demonstrates RTX's dual efficacy in suppressing autoimmunity and alleviating anemia, potentially through indirect effects on inflammatory pathways and iron metabolism. Early RTX use may reduce transfusion dependency and help stabilize renal function in refractory AAV, though advanced fibrosis limits recovery. These findings support RTX as a first-line option in AAV patients with severe anemia and evolving renal injury.
Keywords: ANCA-associated vasculitis, rituximab, Refractory anemia, hepcidin, case report
Received: 26 Mar 2025; Accepted: 04 Aug 2025.
Copyright: © 2025 Shao, Chai, Bai and Luo. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
* Correspondence:
Ningjun Shao, Ningbo No.2 hospital, Ningbo, Zhejiang Province, China
Qun Luo, Ningbo No.2 hospital, Ningbo, Zhejiang Province, China
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