Modern rheumatology case reports最新文献_第6页

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxae083

Shion Kachi, Hideki Oka, Shuji Sumitomo, Shigeo Hara, Koichiro Ohmura

{"title":"IgG4-related disease with epithelioid granulomas: A case and a review of the literature.","authors":"Shion Kachi, Hideki Oka, Shuji Sumitomo, Shigeo Hara, Koichiro Ohmura","doi":"10.1093/mrcr/rxae083","DOIUrl":"10.1093/mrcr/rxae083","url":null,"abstract":"IgG4-related disease (IgG4-RD) is a systemic, immune-mediated, fibroinflammatory disorder that affects multiple organs. Histopathologically, the supportive findings of IgG4-RD include dense lymphocytic infiltrates, obliterative phlebitis, storiform fibrosis, and elevated numbers of IgG4-positive plasma cells. However, the presence of granulomatous inflammation is generally considered highly atypical, suggesting alternative diagnoses such as sarcoidosis and lymphoma. Here, we present a case of IgG4-RD involving granulomatous lymphadenopathy. Labial salivary gland biopsy findings were consistent with IgG4-related sialadenitis. Elevated serum IgG4 levels, hypocomplementemia, and abnormal imaging findings in the kidneys and pancreas indicated an association with IgG4-RD. The patient was treated with prednisolone, which resulted in a significant improvement in the serum IgG4 and complement levels and a notable reduction in lymph node swelling. Although granulomatous inflammation is rare, integrating clinical, serological, radiological, and pathological parameters can ensure an accurate assessment within the appropriate clinicopathological context.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142819734","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

Real-world use of upadacitinib in refractory giant cell arteritis: a case report. Upadacitinib在难治性巨细胞动脉炎中的实际应用：1例报告。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf050

Lisa Wang, Christopher Davidson, Brian Wu, Akihiro Nakamura

{"title":"Real-world use of upadacitinib in refractory giant cell arteritis: a case report.","authors":"Lisa Wang, Christopher Davidson, Brian Wu, Akihiro Nakamura","doi":"10.1093/mrcr/rxaf050","DOIUrl":"10.1093/mrcr/rxaf050","url":null,"abstract":"Giant cell arteritis (GCA) is an autoimmune disease that predominantly affects individuals over 50 years of age by causing inflammation typically in the temporal and cranial arteries. While glucocorticoids like prednisone are the first-line treatment for GCA, glucocorticoid monotherapy is often inadequate for preventing disease flares and is associated with significant side effects when long-term use is required, necessitating the exploration of alternative therapies. Tocilizumab (TCZ) has proven effective as a steroid-sparing agent; however, some patients may respond inadequately or develop adverse events. Treatment with the Janus kinase (JAK) inhibitor upadacitinib (UPA) has recently emerged as a potential alternative therapy for refractory GCA, and its phase III clinical trials successfully demonstrated its efficacy for GCA patients, with or without prior treatment with TCZ. It has also been recently approved by both the European Commission and the U.S. Food and Drug Administration for GCA. However, real-world data on the efficacy of UPA in GCA remain scarce. This case report describes an 82-year-old woman with GCA refractory to both prednisone and TCZ, who reported severe side effects and decreased quality of life from the latter medication. Treatment with UPA resulted in substantial improvements in symptoms, including headaches and fatigue, with minimal negative responses. This outcome demonstrates the potential of UPA as a promising treatment option for GCA patients who are unresponsive or intolerant to current standard therapies. Further real-world studies are warranted to validate UPA's long-term safety and efficacy in treating GCA.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144769482","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

Management of a left anterior descending artery aneurysm in Behçet's disease: A case report. behet病左前降动脉瘤的治疗：1例报告。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf003

Ahmed Hussein Subki, Abdurahman Albeity, Israa Mohammed Mulla, Nabeel Hashim Ismaeil, Muhannad Basheer Qarah, Hussein Halabi

引用次数: 0

Breakthrough herpes zoster following recombinant zoster vaccinations in a rheumatoid arthritis patient receiving a Janus kinase inhibitor: A case report and literature review. 在接受Janus激酶抑制剂的类风湿性关节炎患者中，重组带状疱疹疫苗接种后出现突破性带状疱疹：一例报告和文献综述。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf012

Shunya Nagata, Naoto Yokogawa

{"title":"Breakthrough herpes zoster following recombinant zoster vaccinations in a rheumatoid arthritis patient receiving a Janus kinase inhibitor: A case report and literature review.","authors":"Shunya Nagata, Naoto Yokogawa","doi":"10.1093/mrcr/rxaf012","DOIUrl":"10.1093/mrcr/rxaf012","url":null,"abstract":"The recombinant zoster vaccine (RZV) is immunologically and clinically effective in immunosuppressed patients. Though rheumatoid arthritis (RA) and the Janus kinase inhibitor (JAKi) increase the risk of herpes zoster (HZ) infection, breakthrough cases in which a HZ infection followed RZV administration are rare. We report herein a 63-year-old female patient with seropositive RA who experienced a HZ infection despite receiving the RZV. She had been receiving tocilizumab, methotrexate, and low-dose prednisolone until tocilizumab was switched to upadacitinib 4 weeks after two RZV administrations, which resulted in 63 weeks' remission. Her current admission was for a painful rash consisting of blisters and erythema on the right nasal alar and lips corresponding to the right V2 segment of the trigeminal nerve. HZ was diagnosed and treated for 7 days with intravenous acyclovir, which alleviated the symptoms. JAKi can suppress a range of immunogenic mechanisms which underlie the efficacy of the RZV. The present patient was expected to respond favourably to the RZV because JAKi had not been administered prior to the vaccinations; however, the later start of JAKi therapy caused a breakthrough HZ infection. Immunocompromised patients have a higher risk of severe HZ, including the disseminated form, but breakthrough cases are relatively rare. The RZV is recommended as prophylaxis against HZ as well as means of mitigating its severity when it does occur.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143124196","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

Systemic sclerosis complicated by azathioprine-induced iatrogenic immunodeficiency-associated lymphoproliferative disorder: A case report. 系统性硬化症合并硫唑嘌呤诱导的医源性免疫缺陷相关淋巴细胞增生性疾病1例报告。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf017

Ryota Okazaki, Genki Inui, Yoshihiro Funaki, Miyu Nishigami, Hiroki Kohno, Miki Takata, Tomoya Harada, Akira Yamasaki

{"title":"Systemic sclerosis complicated by azathioprine-induced iatrogenic immunodeficiency-associated lymphoproliferative disorder: A case report.","authors":"Ryota Okazaki, Genki Inui, Yoshihiro Funaki, Miyu Nishigami, Hiroki Kohno, Miki Takata, Tomoya Harada, Akira Yamasaki","doi":"10.1093/mrcr/rxaf017","DOIUrl":"10.1093/mrcr/rxaf017","url":null,"abstract":"Lymphoproliferative disorders are rare complications in patients with autoimmune diseases who are receiving immunosuppressive therapy. This case report describes a 74-year-old man with diffuse cutaneous systemic sclerosis (SSc), anti-RNA polymerase III antibodies, and interstitial pneumonia. The patient's condition initially improved with prednisolone and intravenous cyclophosphamide, followed by maintenance therapy with azathioprine (AZA), nintedanib, and macitentan for pulmonary hypertension. Thirty months after initiating AZA, the patient developed nodules and ulcers in the left lower jaw and philtrum. Skin biopsy confirmed diffuse large B-cell lymphoma. Discontinuation of AZA led to the resolution of the ulcers, and no other lesions were found. This case highlights the risk of iatrogenic immunodeficiency-associated lymphoproliferative disorders in patients with SSc, particularly in those with anti-RNA polymerase III antibodies, who are known to have an increased risk of malignancy. Although methotrexate-associated lymphoproliferative disorders are well documented in patients with rheumatoid arthritis, this is the first reported case of AZA-associated lymphoproliferative disorder in SSc. These findings emphasise the importance of close monitoring of malignancies, including lymphoproliferative disorders, in patients with SSc undergoing immunosuppressive therapy.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143569240","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 : 2025-07-25 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":"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 was resolved with glucocorticoid pulse therapy, high-dose glucocorticoids, and cyclosporine A. However, AOSD relapsed during glucocorticoid tapering. Methotrexate, cyclosporine A, 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":"2025-07-25","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

Anti-neutrophil cytoplasm antibody-negative eosinophilic granulomatous polyangiitis complicated by myositis: a case report. 抗中性粒细胞胞浆抗体阴性嗜酸性肉芽肿性多血管炎合并肌炎1例。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf032

Naoki Nakagawa, Eiichi Kakehi, Kazuhiko Kotani

{"title":"Anti-neutrophil cytoplasm antibody-negative eosinophilic granulomatous polyangiitis complicated by myositis: a case report.","authors":"Naoki Nakagawa, Eiichi Kakehi, Kazuhiko Kotani","doi":"10.1093/mrcr/rxaf032","DOIUrl":"10.1093/mrcr/rxaf032","url":null,"abstract":"A 31-year-old woman visited our hospital with swelling and pain in both forearms of 2 months' duration, followed by swelling and pain in both thighs. Her medical history included bronchial asthma at the age of 18 years. After the birth of her first child at 30 years of age, her asthma worsened and was accompanied by abdominal pain and skin rash, with no identifiable cause. Blood testing showed eosinophilia and high muscle enzyme activities, but she was anti-neutrophilic cytoplasmic antibody (ANCA)-negative. Magnetic resonance imaging of her forearms and thighs revealed strong signals on T2-weighted images in the fascia and muscle. Skin-to-muscle en bloc biopsy showed eosinophilic infiltration of muscle and small vessels. She was diagnosed with eosinophilic granulomatosis with polyangiitis (EGPA), complicated by myositis, although EGPA is usually accompanied by ANCA-positivity in approximately half of cases. Treatment was started with prednisolone alone at 0.5 mg/kg/day, and her symptoms and eosinophil count quickly improved. Clinicians should note the possibility of ANCA-negative EGPA complicated by myositis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144201197","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

Sudden visual loss and hypereosinophilia: A case of eosinophilic granulomatosis with polyangiitis. 突然视力丧失和嗜酸性粒细胞增多：嗜酸性粒细胞肉芽肿病合并多血管炎1例。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf056

Angelo Aita, Raffaella Tiziana Benedetto, Benedetta Goletti, Maria Giovinale, Antonella Velardi, Maria Livia Burzo

{"title":"Sudden visual loss and hypereosinophilia: A case of eosinophilic granulomatosis with polyangiitis.","authors":"Angelo Aita, Raffaella Tiziana Benedetto, Benedetta Goletti, Maria Giovinale, Antonella Velardi, Maria Livia Burzo","doi":"10.1093/mrcr/rxaf056","DOIUrl":"10.1093/mrcr/rxaf056","url":null,"abstract":"Eosinophilic granulomatosis with polyangiitis is a systemic vasculitis of small- and medium-sized blood vessels. The disease can manifest itself variably, with the most commonly affected organs including the lungs, sinuses, and peripheral nervous system. Ocular involvement is rare, and the visual prognosis is generally poor. To date, only a few cases have been published describing the ocular manifestations of eosinophilic granulomatosis with polyangiitis. Given the rarity of these complications, diagnosis can be difficult. We report the case of a 60-year-old woman with a history of asthma, sinusitis, and peripheral neuropathy, who presented to our hospital with sudden loss of vision in her right eye. After referral to an ophthalmologist, a diagnosis of central retinal artery occlusion of the right eye was made. Laboratory tests showed hypereosinophilia and mild positivity for antinuclear antibodies. Imaging revealed multiple micronodules in the lung and sinusopathy. Diagnostic tests for stroke, malignancy, and infectious diseases were negative. Based on laboratory, clinical, and imaging data, the patient was diagnosed with eosinophilic granulomatosis with polyangiitis. Treatment with glucocorticoids and cyclophosphamide was started to induce disease remission. The patient achieved a clinical response to treatment with sustained normalisation of peripheral eosinophil counts and maintenance therapy with mepolizumab was initiated. Unfortunately, no improvement in visual function was observed. In patients with sudden vision loss and hypereosinophilia, eosinophilic granulomatosis with polyangiitis should be suspected. Timely diagnosis is essential to initiate appropriate treatment. However, the effect of systemic treatment on improving patients' visual function is still unclear.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145093342","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

SAPHO syndrome with bone destruction in the lumbar vertebral endplates: A case report. SAPHO综合征伴腰椎终板骨破坏1例报告。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf013

Shiho Nakano, Arata Nakajima, Masato Sonobe, Shinji Taniguchi, Manabu Yamada, Keiichiro Yamamoto, Yasuchika Aoki, Koichi Nakagawa

{"title":"SAPHO syndrome with bone destruction in the lumbar vertebral endplates: A case report.","authors":"Shiho Nakano, Arata Nakajima, Masato Sonobe, Shinji Taniguchi, Manabu Yamada, Keiichiro Yamamoto, Yasuchika Aoki, Koichi Nakagawa","doi":"10.1093/mrcr/rxaf013","DOIUrl":"10.1093/mrcr/rxaf013","url":null,"abstract":"SAPHO syndrome is a rare inflammatory osteoarticular disorder, which includes autoimmune diseases such as pustulotic arthro-osteitis, inflammatory bowel disease-associated spondyloarthritis, and psoriatic arthritis. There are few reports on the treatment of SAPHO syndrome that presents with bone destruction in the spine. We present a case in which adalimumab (ADA) was administered to treat destruction of the lumbar vertebral endplates caused by SAPHO syndrome. The patient was a woman in her 20s who was referred to Toho University Sakura Medical Center with complaints of low back pain; acne on the face, anterior chest, and back; and sternoclavicular joint pain. Blood tests showed a mild increase in C-reactive protein but negative results for rheumatoid factor and anti-cyclic citrullinated peptide antibody. Radiographs and computed tomography images demonstrated destruction with surrounding bone sclerosis in the cranial endplates of the L4 and L5 vertebrae and the left sternoclavicular joint. The Ankylosing Spondylitis Disease Activity Score was 2.05, and the Bath Ankylosing Spondylitis Functional Index was 3.00. Despite the use of the maximum dose of nonsteroidal anti-inflammatory drugs, her symptoms did not improve, and ADA was administered at a dose of 40 mg every 2 weeks. After ADA administration, both the Ankylosing Spondylitis Disease Activity Score and the Bath Ankylosing Spondylitis Functional Index were immediately reduced and low disease activity or remission was maintained thereafter. After 3 years, the computed tomography images showed no progression of bone destruction in the lumbar vertebrae and sternoclavicular joint, and the patient was completely free from low back pain and was able to run normally.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143569269","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

An IL-6 inhibitor subsides iatrogenic aortitis induced by granulocyte-colony stimulating factor without interruption of chemotherapy. 一种IL-6抑制剂可在不中断化疗的情况下减轻粒细胞集落刺激因子诱导的医源性主动脉炎。

IF 0.9

Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf033

Kanako Shimanuki, Yasushi Kondo, Robert T Nakayama, Hiroshi Takei, Jun Kikuchi, Mitsuhiro Akiyama, Hironari Hanaoka, Yuko Kaneko

引用次数: 0