Belimumab for Immune-Mediated Necrotizing Myopathy Associated With Anti-SRP Antibodies: A Case Report and Retrospective Review of Patients Treated With Anti-B-Cell Therapy in a Single Center and Literature
| Autor: | Geng Yin, Qi-Bing Xie, Xin-Yue Ma, Yun-Ru Tian, Bei-Bei Cui |
|---|---|
| Rok vydání: | 2021 |
| Předmět: |
medicine.medical_specialty
Biopsy immune-mediated necrotizing myopathy Immunology Case Report Physical examination Antibodies Monoclonal Humanized Single Center Necrosis rituximab Muscular Diseases Refractory Internal medicine Humans refractory IMNM Immunology and Allergy Medicine Muscle Skeletal B-cell activating factor Glucocorticoids B cell Autoantibodies Retrospective Studies B-Lymphocytes medicine.diagnostic_test biology business.industry SRP antibody RC581-607 Middle Aged Belimumab Treatment Outcome medicine.anatomical_structure biology.protein BAFF Female Rituximab Immunologic diseases. Allergy Antibody belimumab business Signal Recognition Particle Immunosuppressive Agents medicine.drug |
| Zdroj: | Frontiers in Immunology Frontiers in Immunology, Vol 12 (2021) |
| ISSN: | 1664-3224 |
| DOI: | 10.3389/fimmu.2021.777502 |
| Popis: | BackgroundImmune-mediated necrotizing myopathy (IMNM) is characterized by markedly elevated creatinine kinase and histologically scattered necrotic muscle fibers and generally associated with autoantibodies against signal recognition particle (SRP) or 3-hydroxy-3-methylglutaryl-coA-reductase (HMGCR). Poor clinical response to conventional therapies and relapses commonly occur in severe cases. Anti-B-cell therapies have been used in refractory/relapsing cases.MethodsThe characteristics of a patient with IMNM associated with anti-SRP antibodies including physical examination, laboratory tests, and disease activity assessment were evaluated. Conventional therapy, belimumab treatment schedule, and follow-up data were recorded. Medical records of IMNM patients treated in our department from September 2014 to June 2021 were reviewed to evaluate the efficacy and safety of anti-B-cell therapy for anti-SRP IMNM. A literature review of patients with anti-SRP IMNM treated with anti-B-cell therapies was performed.ResultsWe describe a case of a 47-year-old woman with IMNM associated with anti-SRP antibodies who relapsed twice after conventional therapy but showed good response and tolerance to belimumab at 28 weeks follow-up. In this review, three patients from our department were treated with rituximab. Two of the three patients rapidly improved after treatment. Twenty patients and five retrospective studies were included in the literature review. All patients were administered rituximab as an anti-B-cell drug.ConclusionDespite a lack of rigorous clinical trials, considerable experience demonstrated that anti-B-cell therapy might be effective for patients with IMNM associated with anti-SRP antibodies. Belimumab in association with steroids might be an encouraging option for refractory/relapsing cases. |
| Databáze: | OpenAIRE |
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