Modern rheumatology case reports最新文献

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A Unique Case of Granulomatosis with Polyangiitis Presenting with Proximal Muscle Weakness. 一例独特的肉芽肿病合并多血管炎,表现为近端肌肉无力。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-10-06 DOI: 10.1093/mrcr/rxag095
Jason L Chen, Kaitlyn Baber, Jared Stillman, Xiaoshan Gong, Natasha Khona, Trevor Huff, Saher Khalid, Faryal Razzaq, Carlos Tafich-Rios, Hasan Ozgur, Bujji Ainapurapu
{"title":"A Unique Case of Granulomatosis with Polyangiitis Presenting with Proximal Muscle Weakness.","authors":"Jason L Chen, Kaitlyn Baber, Jared Stillman, Xiaoshan Gong, Natasha Khona, Trevor Huff, Saher Khalid, Faryal Razzaq, Carlos Tafich-Rios, Hasan Ozgur, Bujji Ainapurapu","doi":"10.1093/mrcr/rxag095","DOIUrl":"https://doi.org/10.1093/mrcr/rxag095","url":null,"abstract":"<p><p>Granulomatosis with polyangiitis (GPA) is a subtype of ANCA-associated vasculitis that can have a heterogeneous clinical presentation. Although classically involving the respiratory tract and kidneys, GPA can rarely present with predominant skeletal muscle involvement. This is unique given that skeletal muscle involvement has predominantly been associated with eosinophilic GPA and microscopic polyangiitis in the literature. Here, we detail a patient who presented with acute, progressive bilateral proximal lower extremity weakness and thigh pain, along with weight loss and transiently elevated creatine kinase and aldolase. The clinical course was complicated by temporally distinct focal cerebral infarctions, alongside findings of rhinosinusitis and multiple pulmonary nodules. Serology was positive for PR3-ANCA antibodies, and magnetic resonance angiography of the brain utilising black blood sequence confirmed central nervous system (CNS) vasculitis. Crucially, targeted biopsies of the right vastus lateralis and lung nodule demonstrated fibrinoid necrosis and necrotizing granulomatous inflammation, respectively. Together, this histopathological evidence strongly suggests the proximal myopathy and CNS infarction as the result of profound, systemic microvascular ischaemia. This case highlights that GPA may initially manifest as muscle weakness. Recognising this atypical presentation is crucial to ensure timely initiation of treatment to reduce disease-associated morbidity and mortality.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149121214","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 use of a Janus-kinase (JAK) inhibitor in chronic chikungunya arthritis refractory to conventional synthetic DMARDs: a case report. Janus-kinase (JAK)抑制剂在慢性基孔肯雅关节炎中对传统合成DMARDs难治的成功应用:1例报告。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-10-05 DOI: 10.1093/mrcr/rxag098
Jobson Lopes de Oliveira, José Kennedy Amaral, Igor Albuquerque Nogueira, Luis Arthur Brasil Gadelha Farias, Robert T Schoen
{"title":"Successful use of a Janus-kinase (JAK) inhibitor in chronic chikungunya arthritis refractory to conventional synthetic DMARDs: a case report.","authors":"Jobson Lopes de Oliveira, José Kennedy Amaral, Igor Albuquerque Nogueira, Luis Arthur Brasil Gadelha Farias, Robert T Schoen","doi":"10.1093/mrcr/rxag098","DOIUrl":"https://doi.org/10.1093/mrcr/rxag098","url":null,"abstract":"<p><p>Chronic chikungunya arthritis is a debilitating post-viral inflammatory condition that clinically mimics rheumatoid arthritis and often requires long-term treatment with disease-modifying antirheumatic drugs. Treatment options are limited when conventional synthetic disease-modifying antirheumatic drugs are not tolerated or lose efficacy. We report the case of a 50-year-old Brazilian woman with serologically confirmed chikungunya virus infection who developed chronic chikungunya arthritis with a rheumatoid arthritis-like phenotype. Following an acute febrile illness accompanied by rash and negative dengue serology, she was initially managed with symptomatic analgesics, followed by prednisone and hydroxychloroquine. Despite this treatment, the disease activity score in 28 joints calculated with C-reactive protein was elevated at 6.78. Methotrexate was introduced but was discontinued after three weeks due to drug-induced pneumonitis. She later failed leflunomide therapy after 12 months, with a score of 4.33. Baricitinib (4 mg/day), a Janus kinase 1 and 2 inhibitor, was then initiated, resulting in marked clinical and laboratory improvement: the disease activity score decreased to 1.90 over one year, C-reactive protein normalised and the erythrocyte sedimentation rate declined progressively, and morning stiffness resolved, with no infectious complications. This case suggests that Janus kinase inhibition may be a therapeutic option for selected patients with chronic chikungunya arthritis refractory to conventional treatment.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149121406","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
Golimumab-induced lupus erythematosus complicated by angioedema and anti-galactocerebroside antibody-positive dysphagia. 哥利单抗诱导的红斑狼疮并发血管性水肿和抗半乳糖脑苷抗体阳性的吞咽困难。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-10-05 DOI: 10.1093/mrcr/rxag096
Makoto Misaki, Toshiaki Shimizu, Reo Yamazato, Yuichi Suwa, Yoichi Yasunaga, Keishi Fujio
{"title":"Golimumab-induced lupus erythematosus complicated by angioedema and anti-galactocerebroside antibody-positive dysphagia.","authors":"Makoto Misaki, Toshiaki Shimizu, Reo Yamazato, Yuichi Suwa, Yoichi Yasunaga, Keishi Fujio","doi":"10.1093/mrcr/rxag096","DOIUrl":"https://doi.org/10.1093/mrcr/rxag096","url":null,"abstract":"<p><p>Drug-induced lupus erythematosus is an autoimmune condition that resembles systemic lupus erythematosus and occurs after exposure to specific medications, including tumor necrosis factor alpha inhibitors. We report a rare case of golimumab-induced lupus erythematosus complicated by macrophage activation syndrome, angioedema, and severe dysphagia with anti-galactocerebroside antibodies detected. A 57-year-old woman with ulcerative colitis developed facial swelling, malar rash, arthritis, pancytopenia, hyperferritinemia, hypocomplementemia, and proteinuria after long-term treatment with tumor necrosis factor alpha inhibitors, most recently golimumab. Bone marrow examination demonstrated hemophagocytosis, confirming macrophage activation syndrome. Computed tomography and endoscopy revealed soft tissue and esophageal edema consistent with angioedema. Despite improvement of systemic inflammation following high-dose glucocorticoids and intravenous cyclophosphamide, severe dysphagia persisted. A videofluoroscopic swallowing study demonstrated pharyngeal muscle dysfunction, and serum testing detected anti-galactocerebroside immunoglobulin G antibodies. Neurological imaging and electrophysiological studies were unremarkable. Swallowing function gradually recovered after discontinuation of golimumab. This case highlights that golimumab-induced lupus erythematosus may trigger multiple autoimmune complications, including macrophage activation syndrome, angioedema, and antibody-associated neuromuscular dysphagia. Awareness of these potentially life-threatening manifestations is essential for early recognition and appropriate management in patients receiving tumor necrosis factor alpha inhibitors.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149121236","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
Sjögren disease and idiopathic inflammatory myopathy overlap presenting as complement-mediated thrombotic microangiopathy/atypical haemolytic uremic syndrome with a complement factor B variant: A case report. Sjögren疾病和特发性炎性肌病重叠,表现为补体介导的血栓性微血管病/补体因子B变异的非典型溶血性尿毒症综合征:1例报告。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-10-03 DOI: 10.1093/mrcr/rxag097
Momoka Hasegawa, Yoshia Miyawaki, Moe Sakamoto-Tokunaga, Masaki Matoba, Megumi Maeda, Tomokazu Tamura, Natsuki Kubota, Yuya Terajima, Kei Hirose, Kazuya Matsumoto, Manami Watanabe, Takato Nakadoi, Shoichi Nawachi, Keigo Hayashi, Haruki Watanaebe, Eri Katsuyama, Takayuki Katsuyama, Mariko Narasaki, Yoshinori Matsumoto, Noboru Asada, Noritoshi Kato, Jun Wada
{"title":"Sjögren disease and idiopathic inflammatory myopathy overlap presenting as complement-mediated thrombotic microangiopathy/atypical haemolytic uremic syndrome with a complement factor B variant: A case report.","authors":"Momoka Hasegawa, Yoshia Miyawaki, Moe Sakamoto-Tokunaga, Masaki Matoba, Megumi Maeda, Tomokazu Tamura, Natsuki Kubota, Yuya Terajima, Kei Hirose, Kazuya Matsumoto, Manami Watanabe, Takato Nakadoi, Shoichi Nawachi, Keigo Hayashi, Haruki Watanaebe, Eri Katsuyama, Takayuki Katsuyama, Mariko Narasaki, Yoshinori Matsumoto, Noboru Asada, Noritoshi Kato, Jun Wada","doi":"10.1093/mrcr/rxag097","DOIUrl":"https://doi.org/10.1093/mrcr/rxag097","url":null,"abstract":"<p><p>Complement-mediated thrombotic microangiopathy (CM-TMA) is often triggered by autoimmune diseases; however, reports of CM-TMA in patients with Sjögren disease (SjD) and idiopathic inflammatory myopathy (IIM) are limited. We describe the case of a 48-year-old woman with overlapping SjD and IIM who presented with interstitial lung disease. After initiation of combination therapy with calcineurin inhibitor, intravenous cyclophosphamide, and glucocorticoids, she developed progressive thrombocytopenia with schistocytosis, haemolytic anaemia, and acute renal dysfunction. Laboratory testing revealed preserved disintegrin-like and metalloproteinase with thrombospondin type 1 motifs 13 (ADAMTS13) activity (49%) with negative ADAMTS13 inhibitor and Shiga toxins. Despite therapeutic plasma infusion and plasma exchange, she developed severe cardiac dysfunction and abdominal pain, with progressive haematologic and renal dysfunctions requiring haemodialysis. Based on a clinical diagnosis of CM-TMA, eculizumab was initiated, resulting in gradual improvement in haemolysis markers and platelet counts. However, she developed severe multiorgan complications, including cerebellar haemorrhage, acute pancreatitis and coagulopathy, and ultimately died. Genetic testing performed during the clinical course identified a complement factor B (CFB) p.Arg74His variant. This case represents an instance of CM-TMA associated with a SjD/IIM overlap and a CFB variant of uncertain pathogenic significance, suggesting that multiple converging factors may contribute to CM-TMA development.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149121376","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
Anti-c-Mpl antibody-positive acquired amegakaryocytic thrombocytopenia with immune thrombocytopenia-like features in mixed connective tissue disease. 抗c- mpl抗体阳性获得性单核细胞血小板减少伴免疫性血小板减少样特征的混合性结缔组织病
IF 0.9
Modern rheumatology case reports Pub Date : 2026-10-01 DOI: 10.1093/mrcr/rxag093
Tatsuya Ueno, Shunjiro Kurihara, Kodai Kato, Shuhei Kameda, Hiroshi Nakajima
{"title":"Anti-c-Mpl antibody-positive acquired amegakaryocytic thrombocytopenia with immune thrombocytopenia-like features in mixed connective tissue disease.","authors":"Tatsuya Ueno, Shunjiro Kurihara, Kodai Kato, Shuhei Kameda, Hiroshi Nakajima","doi":"10.1093/mrcr/rxag093","DOIUrl":"https://doi.org/10.1093/mrcr/rxag093","url":null,"abstract":"<p><p>Acquired amegakaryocytic thrombocytopenia is a rare cause of severe thrombocytopenia characterized by a marked reduction in bone marrow megakaryocytes and may be difficult to distinguish clinically from immune thrombocytopenia. Acquired amegakaryocytic thrombocytopenia has been reported in various autoimmune and non-autoimmune conditions but has not previously been described in a patient with mixed connective tissue disease. We describe the case of a 63-year-old woman with longstanding mixed connective tissue disease who developed severe thrombocytopenia with mucosal bleeding and purpura. Although she was initially treated for presumed immune thrombocytopenia with platelet transfusion, prednisolone, and intravenous immunoglobulin, followed by intravenous methylprednisolone pulse therapy, her platelet count failed to improve. A subsequent bone marrow biopsy revealed relatively preserved erythroid and granulocytic haematopoiesis with a marked reduction in megakaryocytes, and acquired amegakaryocytic thrombocytopenia was diagnosed after myelodysplastic syndrome and leukaemia had been excluded. Treatment with rituximab and eltrombopag resulted in rapid platelet recovery, and remission has been sustained for 14 months, including after two maintenance rituximab doses administered during follow-up. Testing performed during follow-up revealed positivity for an antibody against the thrombopoietin receptor, suggesting a possible immune-mediated mechanism of impaired megakaryopoiesis. The markedly elevated immature platelet fraction and platelet-associated immunoglobulin G also raised the possibility of concomitant immune thrombocytopenia-like peripheral platelet destruction. This case highlights the importance of considering acquired amegakaryocytic thrombocytopenia in patients with systemic autoimmune disease and severe thrombocytopenia refractory to corticosteroids or intravenous immunoglobulin, while recognizing that impaired platelet production and peripheral platelet destruction may overlap.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149121258","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
Tocilizumab for refractory cerebral arteritis in a patient with systemic lupus erythematosus: A case report. 托珠单抗治疗难治性脑动脉炎1例系统性红斑狼疮:1例报告。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-09-30 DOI: 10.1093/mrcr/rxag094
Yasuto Minegishi, Takehisa Ogura, Chihiro Imaizumi, Reina Maezawa, Takafumi Uchi, Hideto Kameda
{"title":"Tocilizumab for refractory cerebral arteritis in a patient with systemic lupus erythematosus: A case report.","authors":"Yasuto Minegishi, Takehisa Ogura, Chihiro Imaizumi, Reina Maezawa, Takafumi Uchi, Hideto Kameda","doi":"10.1093/mrcr/rxag094","DOIUrl":"https://doi.org/10.1093/mrcr/rxag094","url":null,"abstract":"<p><p>Cerebral arteritis is a rare (<1%) but life-threatening manifestation of systemic lupus erythematosus (SLE). The optimal treatment of cerebral arteritis in refractory cases remains unclear; conventional immunosuppressive therapy results in improvement in arterial stenosis in only 29% of reported cases. We report a 51-year-old woman with a 20-year history of SLE and rheumatoid arthritis who developed aphasia and right hemiparesis due to haemodynamic cerebral infarction caused by cerebral arteritis. Despite treatment with high-dose glucocorticoids, intravenous cyclophosphamide, mycophenolate mofetil, and anifrolumab, the disease remained progressive with persistently elevated C-reactive protein (CRP) levels and worsening arterial stenosis on magnetic resonance angiography (MRA). Cerebrospinal fluid interleukin-6 (IL-6) level was markedly elevated (1590 pg/mL). After switching from anifrolumab to tocilizumab (TCZ), CRP levels normalised, the patient's neurological condition stabilised, and follow-up MRA suggested improvement in arterial stenosis. TCZ might be a viable treatment option for refractory cerebral arteritis in patients with SLE, particularly in cases of elevated cerebrospinal fluid IL-6 levels. To our knowledge, this is the first reported case suggesting the potential efficacy of TCZ in SLE-related cerebral arteritis.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-09-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149083385","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 salvage treatment with baricitinib for macrophage activation syndrome complicating adult-onset Still's disease during interleukin-6 inhibition: A case report and literature review. baricitinib对白细胞介素-6抑制期间巨噬细胞激活综合征并发成人发病斯蒂尔氏病的成功挽救治疗:1例报告和文献综述
IF 0.9
Modern rheumatology case reports Pub Date : 2026-06-29 DOI: 10.1093/mrcr/rxag072
Kazuma Nishisaka, Yo Ueda, Iku Shirasugi, Hirotaka Yamada, Takaichi Okano, Keisuke Nishimura, Sho Sendo, Jun Saegusa
{"title":"Successful salvage treatment with baricitinib for macrophage activation syndrome complicating adult-onset Still's disease during interleukin-6 inhibition: A case report and literature review.","authors":"Kazuma Nishisaka, Yo Ueda, Iku Shirasugi, Hirotaka Yamada, Takaichi Okano, Keisuke Nishimura, Sho Sendo, Jun Saegusa","doi":"10.1093/mrcr/rxag072","DOIUrl":"10.1093/mrcr/rxag072","url":null,"abstract":"<p><p>Adult-onset Still's disease (AOSD) is a systemic autoinflammatory disorder, and macrophage activation syndrome is a life-threatening hyperinflammatory complication. Interleukin-1 and interleukin-6 inhibitors have improved outcomes in refractory AOSD, but optimal management of macrophage activation syndrome, particularly when it develops during biologic therapy, remains uncertain. We report a case of a 49-year-old woman with articular-predominant AOSD who developed fulminant macrophage activation syndrome while receiving high-dose glucocorticoids, tacrolimus, and the interleukin-6 receptor inhibitor tocilizumab. At the onset of macrophage activation syndrome, she had persistent fever, cytopenia, hyperferritinemia, and liver dysfunction, and bone marrow examination revealed haemophagocytosis. Macrophage activation syndrome persisted despite two courses of intravenous methylprednisolone pulse therapy and continuation of tocilizumab. Tocilizumab was discontinued, and treatment was switched to the oral Janus kinase 1/2 inhibitor baricitinib in combination with glucocorticoids and tacrolimus. After this change, the patient experienced rapid defervescence, marked improvement in blood counts and ferritin levels, and sustained control of articular and systemic disease activity. Glucocorticoids were successfully tapered without relapse, and the patient remained in remission for more than 2 years without serious infections. To contextualise this case, we reviewed published reports on Janus kinase inhibition in AOSD, including cases complicated by macrophage activation syndrome. Multi-cytokine blockade through Janus kinase 1/2 inhibition, targeting overlapping interleukin-6, interferon-gamma, and granulocyte-macrophage colony-stimulating factor pathways, may simultaneously suppress macrophage activation and systemic inflammation. This case highlights the potential of baricitinib as a therapeutic option for refractory AOSD-associated macrophage activation syndrome during interleukin-6 inhibition.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148427617","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
Skin vasculitis and cardiac eosinophil extracellular trap formation in eosinophilic granulomatosis with polyangiitis: A case report. 嗜酸性肉芽肿合并多血管炎并发皮肤血管炎和心脏嗜酸性粒细胞胞外陷阱形成1例。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-06-29 DOI: 10.1093/mrcr/rxag046
Hisashi Kamido, Yuki Oba, Masayuki Yamanouchi, Tatsuya Suwabe, Kei Kono, Kenichi Ohashi, Keinosuke Hizuka, Shigeharu Ueki, Naoki Sawa, Yoshifumi Ubara
{"title":"Skin vasculitis and cardiac eosinophil extracellular trap formation in eosinophilic granulomatosis with polyangiitis: A case report.","authors":"Hisashi Kamido, Yuki Oba, Masayuki Yamanouchi, Tatsuya Suwabe, Kei Kono, Kenichi Ohashi, Keinosuke Hizuka, Shigeharu Ueki, Naoki Sawa, Yoshifumi Ubara","doi":"10.1093/mrcr/rxag046","DOIUrl":"10.1093/mrcr/rxag046","url":null,"abstract":"<p><p>The pathophysiology of organ involvement in eosinophilic granulomatosis with polyangiitis is characterised by vasculitis and eosinophilic inflammation, which can be accompanied by eosinophil extracellular trap cell death (EETosis). Clinically, eosinophilic inflammation is often observed in the lungs, gastrointestinal tract, and heart, whereas vasculitis more frequently affects the peripheral nerves, skin, and kidneys. However, histopathological confirmation of vasculitis and EETosis in different organs within the same patient has rarely been reported. We report a patient with EGPA presenting with both cutaneous and cardiac involvement. Skin biopsy findings were consistent with cutaneous fibrinoid vasculitis with prominent eosinophilic infiltration. Prednisolone therapy led to improvement of the skin lesion and inflammatory markers within 1 month. In contrast, endomyocardial biopsy demonstrated marked eosinophilic infiltration with cytolytic degranulation and extracellular deposition of eosinophil granule proteins, without evidence of fibrinoid vasculitis. Immunofluorescence findings supported the presence of EETosis. Despite prompt methylprednisolone pulse therapy, the cardiac lesion showed limited response and was complicated by refractory arrhythmias. This case suggests that distinct pathogenic mechanisms, namely vasculitis and EETosis, may predominate in different organs even within the same patient, potentially leading to discrepant treatment responses. When EETosis-driven pathology is dominant, conventional immunosuppressive therapy may have limited efficacy, highlighting the need for therapeutic strategies targeting EETosis.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148311074","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
Reconstruction preserving the talonavicular and subtalar joints for flexible rocker-bottom foot deformity in rheumatoid arthritis. 保留距舟关节和距下关节的重建治疗类风湿性关节炎的柔性摇底足畸形:1例报告。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-06-29 DOI: 10.1093/mrcr/rxag066
Takuya Kurihara, Takaaki Noguchi, Yuki Etani, Atsushi Sugimoto, Seiji Okada, Kosuke Ebina
{"title":"Reconstruction preserving the talonavicular and subtalar joints for flexible rocker-bottom foot deformity in rheumatoid arthritis.","authors":"Takuya Kurihara, Takaaki Noguchi, Yuki Etani, Atsushi Sugimoto, Seiji Okada, Kosuke Ebina","doi":"10.1093/mrcr/rxag066","DOIUrl":"10.1093/mrcr/rxag066","url":null,"abstract":"<p><p>A patient with rheumatoid arthritis and a flexible rocker-bottom foot deformity underwent surgery, including a flexor digitorum longus transfer, a spring ligament reconstruction, and a lateral column lengthening using calcaneocuboid distraction arthrodesis, while preserving the talonavicular and subtalar joints. Correction was achieved with the restoration of the medial longitudinal arch, and clinical outcome scores had improved at the final follow-up (1 year postoperatively). This case suggests that, even in patients with inflammatory joint diseases presenting with a rocker-bottom foot deformity, reconstruction preserving the talonavicular and subtalar joints may be considered as a treatment option when disease activity is adequately controlled with pharmacotherapy and the talonavicular and subtalar joints are not destroyed, as in the present case.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148407554","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
Salvage surgery for talar component subsidence after total ankle arthroplasty using the FINE Total Ankle System: A case report. 全踝关节置换术后使用FINE全踝关节系统进行距骨构件沉降的抢救手术一例报告。
IF 0.9
Modern rheumatology case reports Pub Date : 2026-06-29 DOI: 10.1093/mrcr/rxag058
Takaaki Noguchi, Yuki Etani, Gensuke Okamura, Makoto Hirao, Jun Hashimoto, Atsushi Sugimoto, Seiji Okada, Kosuke Ebina
{"title":"Salvage surgery for talar component subsidence after total ankle arthroplasty using the FINE Total Ankle System: A case report.","authors":"Takaaki Noguchi, Yuki Etani, Gensuke Okamura, Makoto Hirao, Jun Hashimoto, Atsushi Sugimoto, Seiji Okada, Kosuke Ebina","doi":"10.1093/mrcr/rxag058","DOIUrl":"10.1093/mrcr/rxag058","url":null,"abstract":"<p><p>In recent years, total ankle arthroplasty (TAA) has been increasingly selected for the treatment of ankle arthropathy associated with rheumatoid arthritis because it allows early weight-bearing and preservation of ankle range of motion. However, salvage strategies for talar component subsidence after TAA depend on the implant system used, and ankle arthrodesis is frequently selected for cases treated with the FINE Total Ankle System (NAKASHIMA HEALTHFORCE CO.). Implant removal followed by ankle arthrodesis inevitably results in loss of ankle motion and substantial bone defects. Furthermore, removal of a cemented implant often requires extensive bone debridement, which may further increase the risk of non-union and deterioration of activities of daily living due to prolonged non-weight-bearing. We report a case of talar component subsidence of approximately 10 mm after TAA treated with the FINE Total Ankle System, successfully managed by revision TAA using the largest available talar component (size 6L) combination with bone grafting and medial malleolar osteotomy. This approach preserved ankle range of motion and was associated with favourable mid-term clinical and radiographic outcomes in the present case. Revision TAA using the largest available talar component with bone grafting may be considered a potential salvage option for carefully selected patients with talar component subsidence after TAA using the FINE Total Ankle System.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148427635","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}
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