Modern rheumatology case reports最新文献

A case of irAE myositis with positive anti-striational antibodies after anti-PD-L1 antibody administration: a case report. 抗pd - l1抗体给药后抗条纹抗体阳性的irAE肌炎1例。

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Modern rheumatology case reports Pub Date : 2025-05-30 DOI: 10.1093/mrcr/rxaf025

Hironori Ando, Ken Takao, Makie Honda, Saki Kubota, Tokuyuki Hirose, Takehiro Kato, Masami Mizuno, Takuo Hirota, Yukio Horikawa, Daisuke Yabe

{"title":"A case of irAE myositis with positive anti-striational antibodies after anti-PD-L1 antibody administration: a case report.","authors":"Hironori Ando, Ken Takao, Makie Honda, Saki Kubota, Tokuyuki Hirose, Takehiro Kato, Masami Mizuno, Takuo Hirota, Yukio Horikawa, Daisuke Yabe","doi":"10.1093/mrcr/rxaf025","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf025","url":null,"abstract":"Immune checkpoint inhibitors (ICIs) are widely used in clinical practice, necessitating appropriate management of immune-related adverse events (irAEs). Although severe neurologic irAEs are less common, they often lead to poor outcomes, requiring early detection and prompt intervention. An 88-year-old woman with invasive urothelial carcinoma received 6 cycles of gemcitabine plus carboplatin followed by avelumab, an anti-PD-L1 antibody, as maintenance therapy. One week later, she developed progressive limb weakness and was diagnosed with irAE myositis based on elevated CK levels and imaging findings. Early treatment with methylprednisolone pulse therapy, followed by prednisolone (1 mg/kg BW), led to rapid improvement, and no relapse occurred after PSL completion at four months. irAE myositis has clinical, pathological and immunological features that differ from known inflammatory muscle diseases. In this case, the time to onset and the presence of anti-striational antibodies were consistent with previous reports focusing on anti-PD-1 antibodies, whereas ocular symptoms, myocarditis, and myasthenia gravis, which are considered characteristic of irAE myositis, were not observed. Given the expected increase in high-grade neurological irAEs, accumulating case reports is essential to better understand the differences in clinical presentation and prognosis, which may vary depending on drug-specific effects and autoantibody profiles. Furthermore, this case suggests that some patients with irAE myositis may successfully taper or discontinue prednisolone earlier than traditionally expected.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144188740","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Successful treatment with tocilizumab in a case of familial Mediterranean fever with Takayasu arteritis. 托珠单抗成功治疗家族性地中海热伴高须动脉炎1例。

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Modern rheumatology case reports Pub Date : 2025-05-28 DOI: 10.1093/mrcr/rxaf027

Saeka Kondo, Natsuka Umezawa, Yasuhiro Tagawa, Shinsuke Yasuda

{"title":"Successful treatment with tocilizumab in a case of familial Mediterranean fever with Takayasu arteritis.","authors":"Saeka Kondo, Natsuka Umezawa, Yasuhiro Tagawa, Shinsuke Yasuda","doi":"10.1093/mrcr/rxaf027","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf027","url":null,"abstract":"Familial Mediterranean fever (FMF) is an autoinflammatory disease associated with mutations in MEFV, which encodes pyrin. Patients with FMF present intermittent high fever with elevated inflammatory markers during periodic attacks. While some forms of vasculitis including IgA vasculitis and polyarteritis nodosa have been reported in some patients with FMF, Takayasu arteritis (TAK) rarely associated with FMF. In addition, little has been known about clinical features and pathogenesis of vasculitis with FMF. Here we report a case of FMF with TAK. Our case is remarkable on his clinical course of neck pain with low-grade elevation of serum C-reactive protein during interictal periods of fever attacks. He possessed the dual genetic-background of pathogenic variant of p.M694V in MEFV and HLA-B*52:01 which is susceptible to TAK. Although he was refractory to the combination therapy with colchicine, corticosteroids and methotrexate, tocilizumab was effective for both recurrent fever attacks and vasculitis. Previous four reports of FMF with TAK as well as our case suggest the pathogenic MEFV mutation could be a predisposing or additional factor that modify the development of TAK. Since both the activity of FMF and TAK responded to tocilizumab in our case, the pathogenesis shared between FMF and TAK was indicated.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144164420","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A case of Schnitzler syndrome complicated by rheumatoid arthritis treated with methotrexate. 甲氨蝶呤治疗Schnitzler综合征合并类风湿关节炎1例。

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Modern rheumatology case reports Pub Date : 2025-05-28 DOI: 10.1093/mrcr/rxaf026

Kensuke Irino, Yu Kochi, Sakurako Imamura, Chika Nabeshima, Naoya Oka, Takuya Sawabe

{"title":"A case of Schnitzler syndrome complicated by rheumatoid arthritis treated with methotrexate.","authors":"Kensuke Irino, Yu Kochi, Sakurako Imamura, Chika Nabeshima, Naoya Oka, Takuya Sawabe","doi":"10.1093/mrcr/rxaf026","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf026","url":null,"abstract":"Schnitzler syndrome (SchS) is a rare autoinflammatory disorder characterized by recurrent urticaria, monoclonal gammopathy, a fever, and arthralgia. We herein report a case of SchS complicated by rheumatoid arthritis (RA). A 78-year-old man presented to our hospital with a fever, myalgia, and urticaria, each lasting for approximately 1 week over the past 8 years. He was diagnosed with SchS based on monoclonal IgG gammopathy, chronic urticaria, an intermittent fever, arthritis, high inflammatory markers, and neutrophil infiltration in the dermis on skin biopsy. Although colchicine improved slightly, the patient subsequently developed arthritis and was diagnosed with RA based on elevated anti-cyclic citrullinated peptide antibody and rheumatoid factor levels. Methotrexate (MTX), a first-line therapy for RA, was initiated, resulting in a remarkable improvement in both RA and SchS symptoms. This case highlights the rare coexistence of SchS and RA as well as the efficacy of MTX in treating both conditions. Although interleukin (IL)-1 inhibitors are considered the most effective treatment for SchS, they are not approved for SchS in Japan and are expensive. The efficacy of IL-6 inhibitors in SchS has also been reported, and MTX suppresses inflammatory cytokines including IL-1 and IL-6. As shown in our case, drugs that modulate IL-6 levels, such as MTX, may be a viable treatment option for SchS.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144164399","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Macrophage activation syndrome with hypoplastic marrow and Orbital myositis as a unique initial presentation of Systemic Lupus Erythematosus: A Case-Based Review. 巨噬细胞激活综合征伴骨髓发育不良和眼眶肌炎是系统性红斑狼疮的独特初始表现：一项基于病例的回顾。

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Modern rheumatology case reports Pub Date : 2025-05-28 DOI: 10.1093/mrcr/rxaf028

Kaustav Bhowmick, Srijana Pradhan, Uddalok Das, Arghya Chattopadhyay, Pasang Lhamu Sherpa

{"title":"Macrophage activation syndrome with hypoplastic marrow and Orbital myositis as a unique initial presentation of Systemic Lupus Erythematosus: A Case-Based Review.","authors":"Kaustav Bhowmick, Srijana Pradhan, Uddalok Das, Arghya Chattopadhyay, Pasang Lhamu Sherpa","doi":"10.1093/mrcr/rxaf028","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf028","url":null,"abstract":"Macrophage activation syndrome (MAS) with hypoplastic marrow is a rare, but life-threatening presenting manifestation of systemic lupus erythematosus (SLE). Lupus orbitopathy also is very rarely reported in the literature. We hereby report for the first time a 19-year-old girl who had a unique combination of MAS with hypoplastic marrow and orbitopathy as a presenting feature of lupus. We discussed in detail our patient's demographic characteristics, clinical features, treatment, and outcome. Our patient responded well to high-dose glucocorticoids along with 6 monthly doses of intravenous cyclophosphamide as per the modified National Institute of Health protocol followed by tacrolimus. Her prednisolone dose could be tapered to 7.5 mg daily after eight months of treatment. We made a search on PubMed and Scopus for a literature review. We obtained 15 cases of MAS as a presenting feature in lupus, of which four had associated hypoplastic marrow. Males and juvenile age groups were equally affected with MAS as a presenting feature of lupus. Most of the patients responded to high-dose glucocorticoids. Only one patient succumbed to MAS. We also reviewed 14 cases of lupus orbitopathy with their clinical, and imaging characteristics, treatment, and outcome. Females predominated in these cases with mean age being 43.6 years (SD +/- 16.9 years). Treatment with glucocorticoids and immunosuppressive agents leads to complete resolution in most patients. The diagnosis of MAS with orbitopathy as a presenting feature of lupus in a 19-year-old girl poses a diagnostic challenge and requires the prompt exclusion of the common mimickers before initiation of aggressive immunosuppression.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144164402","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Successful Treatment of Recurrent Hemarthrosis after Total Knee Arthroplasty Using Genicular Artery Embolization in a Patient with Rheumatoid Arthritis. 类风湿性关节炎患者膝动脉栓塞成功治疗全膝关节置换术后复发性关节血肿1例。

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Modern rheumatology case reports Pub Date : 2025-05-28 DOI: 10.1093/mrcr/rxaf030

Mayu Iiboshi, Hiroyasu Mizuhara, Kenta Makabe, Yasunori Omata, Takeyuki Watadani, Sakae Tanaka, Takumi Matsumoto

{"title":"Successful Treatment of Recurrent Hemarthrosis after Total Knee Arthroplasty Using Genicular Artery Embolization in a Patient with Rheumatoid Arthritis.","authors":"Mayu Iiboshi, Hiroyasu Mizuhara, Kenta Makabe, Yasunori Omata, Takeyuki Watadani, Sakae Tanaka, Takumi Matsumoto","doi":"10.1093/mrcr/rxaf030","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf030","url":null,"abstract":"Recurrent hemarthrosis after total knee arthroplasty (TKA) is a rare but challenging complication, with an incidence ranging from 0.3% to 1.6%. Conservative treatment, including rest, joint aspiration, and oral tranexamic acid, is often the first-line approach, but approximately two-thirds of cases require more invasive procedures. Genicular artery embolization (GAE) has emerged as a promising alternative to synovectomy for refractory cases, offering high success rates with fewer complications. While most reports of GAE for recurrent hemarthrosis focus on patients with osteoarthritis, data on its use in patients with rheumatoid arthritis (RA) are scarce. We report a case of a 79-year-old woman with RA who presented with recurrent hemarthrosis 11 years after TKA. Despite conservative treatment, her hemarthrosis persisted. Angiography revealed three hypervascular areas supplied by branches of the superior lateral, superior medial, and middle genicular arteries. Embolization was successfully performed using gelatin sponge, resulting in complete symptom resolution without recurrence at the two-year follow-up. This case highlights the potential of GAE as a viable treatment option for recurrent hemarthrosis in patients with RA, particularly those with multiple bleeding sites or when synovectomy is unsuccessful or impractical. Careful evaluation from multiple angiographic angles is crucial to detect all bleeding sources, especially in post-TKA cases where metal implants may obscure vascular regions. Although further studies are needed, GAE may represent a safe and effective alternative for managing recurrent hemarthrosis in patients with RA.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144164417","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Atypical Lymphoproliferative Disorder and Acquired Angioedema in Systemic Lupus Erythematosus and Sjögren's Syndrome: A Diagnostic Challenge. 系统性红斑狼疮和Sjögren综合征的非典型淋巴增生性疾病和获得性血管性水肿：诊断挑战。

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Modern rheumatology case reports Pub Date : 2025-05-26 DOI: 10.1093/mrcr/rxaf022

Junya Kitai, Takahiro Seno, Masataka Kohno, Keiko Hashimoto, Shinsuke Mizutani, Junya Kuroda, Yasufumi Masaki, Yutaka Kawahito

{"title":"Atypical Lymphoproliferative Disorder and Acquired Angioedema in Systemic Lupus Erythematosus and Sjögren's Syndrome: A Diagnostic Challenge.","authors":"Junya Kitai, Takahiro Seno, Masataka Kohno, Keiko Hashimoto, Shinsuke Mizutani, Junya Kuroda, Yasufumi Masaki, Yutaka Kawahito","doi":"10.1093/mrcr/rxaf022","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf022","url":null,"abstract":"We report a case of a 33-year-old woman presenting with facial erythema, pharyngitis, and progressive cervical lymphadenopathy with tongue, pharyngeal, and laryngeal edema. Laboratory evaluations revealed an elevated soluble interleukin-2 receptor level, positive antinuclear, double-stranded DNA, and Sjögren's syndrome-related antigen A (SS-A) antibodies, and decreased complement levels. Salivary gland studies confirmed Sjögren's syndrome, and together with serological findings, the patient fulfilled criteria for systemic lupus erythematosus (SLE). Although Positron emission tomography-computed tomography (PET-CT) findings raised suspicion for malignant lymphoma, a cervical lymph node biopsy demonstrated interfollicular expansion, proliferation of small blood vessels with prominent endothelial cells, and large immunoblasts, consistent with atypical lymphoplasmacytic and immunoblastic proliferation (ALPIBP). Despite clinical evidence of angioedema on endoscopic examination, complement levels and C1-inhibitor activity were normal, suggesting a non-complement-mediated mechanism. The patient was initially treated with hydroxychloroquine for SLE; however, worsening symptoms required prednisolone therapy, leading to significant clinical improvement. Recurrence during steroid tapering was managed by adjusting the steroid dose and adding mycophenolate mofetil. This case emphasizes the importance of comprehensive evaluation to differentiate ALPIBP from malignant lymphoma and to recognize atypical acquired angioedema in autoimmune disease.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144145334","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A rare large vessels involvement in a patient with a clinical diagnosis of Granulomatosis with polyangiitis: A case report. 临床诊断为肉芽肿病合并多血管炎的罕见大血管受累1例。

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Modern rheumatology case reports Pub Date : 2025-05-26 DOI: 10.1093/mrcr/rxaf021

Mona Terkmani, Yasmin Najjar, Fatema Naddaf, Sana Shaikh Torab, Taher Sawas, Sami Albitar

引用次数: 0

Successful treatment by systemic corticosteroids and intravenous cyclophosphamide in acquired pure red cell aplasia associated with systemic lupus erythematosus. 全身性皮质类固醇和静脉注射环磷酰胺成功治疗与系统性红斑狼疮相关的获得性纯红细胞发育不全。

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Modern rheumatology case reports Pub Date : 2025-05-26 DOI: 10.1093/mrcr/rxaf024

Takafumi Tsushima, Masashi Fukuta, Natsumi Yoda, Chiharu Kimeda, Kazusuke Tanaka, Kosuke Matsuo, Yasuhito Hatanaka, Rena Matsumoto, Sonoko Shimoji, Yoshikazu Utsu, Shin-Ichi Masuda, Nobuyuki Aotsuka

{"title":"Successful treatment by systemic corticosteroids and intravenous cyclophosphamide in acquired pure red cell aplasia associated with systemic lupus erythematosus.","authors":"Takafumi Tsushima, Masashi Fukuta, Natsumi Yoda, Chiharu Kimeda, Kazusuke Tanaka, Kosuke Matsuo, Yasuhito Hatanaka, Rena Matsumoto, Sonoko Shimoji, Yoshikazu Utsu, Shin-Ichi Masuda, Nobuyuki Aotsuka","doi":"10.1093/mrcr/rxaf024","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf024","url":null,"abstract":"Commonly acquired pure red cell aplasia (PRCA) can be associated with an underlying autoimmune disease. Reports of PRCA associated with systemic lupus erythematosus (SLE) are rare, and no treatment strategies have been documented. We report a case of PRCA associated with SLE that responded to intravenous cyclophosphamide (IVCY) and systemic corticosteroids. A 62-year-old Japanese female was diagnosed with PRCA and SLE. At the time of the initial visit, the patient simultaneously presented with severe proteinuria, massive pleural effusion, and interstitial changes. Notably, cyclosporine and corticosteroids did not improve PRCA and SLE status of the patient. After IVCY and corticosteroid administration, the patient's reticulocyte count and anaemia improved. Various other symptoms associated with SLE also improved. Cyclophosphamide is typically administered in PRCA in small, continuous oral doses. However, in this case, we did not need to initiate this continuous, low-dose treatment after IVCY. Overall, this study highlights therapeutic strategies involving IVCY in treating PRCA associated with SLE.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144145357","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

The role of syphilis in exacerbating axial spondyloarthritis symptoms: A case report on diagnosis and management. 梅毒在加重轴型脊柱炎症状中的作用：1例诊断和治疗报告。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-05-26 DOI: 10.1093/mrcr/rxaf023

Junya Kitai, Takahiro Seno, Naoka Kamio, Masataka Kohno, Tomoyuki Shirase, Yutaka Kawahito

{"title":"The role of syphilis in exacerbating axial spondyloarthritis symptoms: A case report on diagnosis and management.","authors":"Junya Kitai, Takahiro Seno, Naoka Kamio, Masataka Kohno, Tomoyuki Shirase, Yutaka Kawahito","doi":"10.1093/mrcr/rxaf023","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf023","url":null,"abstract":"Syphilis, caused by Treponema pallidum, is resurging globally, with diverse clinical manifestations. While musculoskeletal involvement is recognized, its role in exacerbating axial spondyloarthritis (ax-SpA) or causing infectious spondylitis is poorly understood. We report the case of a 48-year-old man with long-standing ax-SpA who experienced spontaneous remission until a severe flare triggered by primary syphilis, characterized by an ulcerative lesion on the upper lip. Imaging studies revealed ankylosis of the sacroiliac joints and new inflammatory changes in the lumbar spine. Specifically, plain radiography demonstrated complete ankylosis of the sacroiliac joints, while MRI revealed bone contrast enhancement, consistent with active inflammation. Syphilis infection was confirmed both serologically and by immunohistochemical staining of tissue samples. Targeted antibiotic therapy with penicillin G and amoxicillin-probenecid led to significant clinical and radiological improvement. This case highlights syphilis as a potential trigger for ax-SpA exacerbation or an infectious spinal process. Recognizing syphilis in patients with unexplained axial inflammation can prevent unnecessary immunosuppressive therapy and guide effective management. With the rising incidence of syphilis, prompt diagnosis and treatment are critical for preventing spinal pathology progression and improving outcomes.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-05-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144145364","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Successful re-administration of tocilizumab in a patient with adult-onset Still's disease after improvement of macrophage activation syndrome. 在巨噬细胞激活综合征改善后，托珠单抗在成人发病斯蒂尔氏病患者中的成功再给药。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-04-10 DOI: 10.1093/mrcr/rxaf020

Yuma Nagasawa, Kaoru Takase-Minegishi, Soichiro Adachi, Kento Ichikawa, Hideto Nagai, Tomoya Watanabe, Yukie Yamaguchi, Ryusuke Yoshimi, Yohei Kirino, Hideaki Nakajima

{"title":"Successful re-administration of tocilizumab in a patient with adult-onset Still's disease after improvement of macrophage activation syndrome.","authors":"Yuma Nagasawa, Kaoru Takase-Minegishi, Soichiro Adachi, Kento Ichikawa, Hideto Nagai, Tomoya Watanabe, Yukie Yamaguchi, Ryusuke Yoshimi, Yohei Kirino, Hideaki Nakajima","doi":"10.1093/mrcr/rxaf020","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf020","url":null,"abstract":"Tocilizumab (TCZ) is effective for inducing remission in adult-onset Still's disease (AOSD), but its use may occasionally trigger macrophage activation syndrome (MAS). The rationale for re-introducing TCZ in patients with a history of MAS is not well established. Here, we report a case of successful re-administration of TCZ for an AOSD relapse in a patient with a prior history of MAS during TCZ therapy. A 67-year-old woman, initially treated with TCZ for polyarthritis, developed MAS associated with AOSD. MAS were resolved with glucocorticoid pulse therapy, high-dose glucocorticoids, and cyclosporine A (CyA). However, AOSD relapsed during glucocorticoid tapering. Methotrexate, CyA, and repeated glucocorticoid pulses failed to control the disease. Following another glucocorticoid pulse, TCZ (8 mg/kg weekly) was re-introduced intravenously. This approach allowed successful glucocorticoid tapering and long-term remission. This case highlights the complexities of managing AOSD: while the initial TCZ therapy may have contributed to the onset of MAS, the subsequent re-introduction of TCZ enabled effective disease control and sustained remission.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144030136","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0