Modern rheumatology case reports最新文献_第4页

Paediatric SAPHO syndrome with pleural effusion: Case report of a unique finding in a rare disease. 伴有胸腔积液的小儿 SAPHO 综合征：罕见疾病中一个独特发现的病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae038

Christopher Kruger, Christine Wang, Andrew Grim

{"title":"Paediatric SAPHO syndrome with pleural effusion: Case report of a unique finding in a rare disease.","authors":"Christopher Kruger, Christine Wang, Andrew Grim","doi":"10.1093/mrcr/rxae038","DOIUrl":"10.1093/mrcr/rxae038","url":null,"abstract":"Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome is a rare autoinflammatory disease characterised by bone inflammation and skin manifestations including acne, palmoplantar pustulosis, psoriasis, or hidradenitis suppurativa. SAPHO syndrome is considered on the same spectrum as chronic nonbacterial osteomyelitis/chronic recurrent multifocal osteomyelitis, the former often being the nomenclature in adults and the latter in children. The diagnosis is made on patterns of clinical manifestations and is a diagnosis of exclusion. While skin and bone manifestations are commonly described with SAPHO syndrome, pleural involvement is rare, and few cases have been described in the literature, especially in paediatric patients. Herein we present a 14-year-old female with a past medical history of hidradenitis supprtiva, eczema, psoriasis, and a prior episode of culture-negative osteomyelitis who presented to the emergency room with chief complaints of right-sided pain with inspiration and back pain. Exam revealed palmoplantar pustulosis, hidradenitis supprativa, psoriasis, and tenderness of vertebrae. Imaging showed a right-sided pleural effusion and multiple sites of osteitis. Laboratory evaluation revealed elevated inflammatory markers, an exudative pleural effusion with neutrophilic predominance, and no evidence of malignancy, infection, or immunodeficiency. The patient was diagnosed with SAPHO syndrome and treated with naproxen, methotrexate, and golimumab with significant improvement including resolution of the pleural effusion. Paediatric SAPHO syndrome is a rare disease that classically causes osteitis and skin manifestations. This case highlights that pleural effusion can be a rare manifestation of paediatric SAPHO syndrome. Patients with suspected SAPHO syndrome with respiratory symptoms should be evaluated for pleural effusion.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"168-173"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141857476","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 vesiculobullous dermatomyositis with anti-NXP-2 antibody without malignancy. 一例无恶性肿瘤的抗 NXP-2 抗体膀胱脓肿性皮肌炎病例

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae037

Ryota Naito, Ryosuke Hiwa, Ryuta Inaba, Kosaku Murakami, Akihiko Kitoh, Yo Kaku, Teruasa Murata, Yuki Ichimura, Naoko Okiyama, Ichizo Nishino, Mirei Shirakashi, Hideo Onizawa, Hideaki Tsuji, Koji Kitagori, Shuji Akizuki, Ran Nakashima, Akira Onishi, Masao Tanaka, Hajime Yoshifuji, Akio Morinobu

{"title":"A case of vesiculobullous dermatomyositis with anti-NXP-2 antibody without malignancy.","authors":"Ryota Naito, Ryosuke Hiwa, Ryuta Inaba, Kosaku Murakami, Akihiko Kitoh, Yo Kaku, Teruasa Murata, Yuki Ichimura, Naoko Okiyama, Ichizo Nishino, Mirei Shirakashi, Hideo Onizawa, Hideaki Tsuji, Koji Kitagori, Shuji Akizuki, Ran Nakashima, Akira Onishi, Masao Tanaka, Hajime Yoshifuji, Akio Morinobu","doi":"10.1093/mrcr/rxae037","DOIUrl":"10.1093/mrcr/rxae037","url":null,"abstract":"Vesiculobullous dermatomyositis (VD) is a rare manifestation of dermatomyositis (DM) and has been suggested to be associated with malignancy. Although the myositis-specific autoantibodies are associated with distinct clinical presentations of DM, those associated with VD remain unclear. Here, we present the case of a 54-year-old man with VD who tested positive for antinuclear matrix protein 2 (NXP-2) antibodies, one of the DM-specific autoantibodies. Serological and histopathological findings did not support autoimmune blistering disease. Physical and histological findings suggested that the severe oedema in combination with the interface dermatitis of DM contributed to blister formation. Although a systemic examination was performed, no evidence of malignancy was found. Following initiation of immunosuppressive therapy, the patient showed significant improvement in both skin lesions and myositis. This case represents the first report of anti-NXP-2-positive VD without malignancy or autoimmune blistering disease. Subcutaneous oedema, a characteristic feature of anti-NXP-2-positive DM, could be related to the formation of VD.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"84-87"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141861955","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

Acute heart failure with reduced ejection fraction following ozoralizumab in a patient with rheumatoid arthritis: A case report. 类风湿性关节炎患者使用奥唑来珠单抗后出现射血分数降低的急性心力衰竭：病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae053

Takaaki Ito, Yuji Miyoshi

引用次数: 0

Mid-term outcomes of distal tibial oblique osteotomy in patients with rheumatoid arthritis: A report of three cases. 类风湿性关节炎患者胫骨远端斜截骨术的中期疗效：三例病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae068

Takaaki Noguchi, Makoto Hirao, Kosuke Ebina, Yuki Etani, Gensuke Okamura, Manabu Sakata, Taihei Miura, Seiji Okada, Jun Hashimoto

引用次数: 0

Persistent liver injury following avacopan discontinuation in antineutrophil cytoplasmic antibody-associated vasculitis: A rare case of liver biopsy in the chronic phase of liver injury. 抗中性粒细胞胞浆抗体相关性血管炎患者停用阿伐戈班后的持续性肝损伤：肝损伤慢性期肝活检的罕见病例。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae071

Hiroyuki Yamaguchi, Nayu Fujii, Hideki Shimizu

{"title":"Persistent liver injury following avacopan discontinuation in antineutrophil cytoplasmic antibody-associated vasculitis: A rare case of liver biopsy in the chronic phase of liver injury.","authors":"Hiroyuki Yamaguchi, Nayu Fujii, Hideki Shimizu","doi":"10.1093/mrcr/rxae071","DOIUrl":"10.1093/mrcr/rxae071","url":null,"abstract":"Drug-induced liver injury is a major concern associated with the use of avacopan, a new therapeutic agent for antineutrophil cytoplasmic antibody-associated vasculitis. Here, we report a rare instance of liver biopsy performed for persistent liver injury following discontinuation of avacopan in an elderly woman with microscopic polyangiitis. The patient developed cholestatic liver injury after 2 months of avacopan therapy, which led to drug cessation. Despite the discontinuation of avacopan followed by supportive care, the liver enzyme levels remained elevated, necessitating liver biopsy. Histological examination revealed residual inflammation and focal necrosis around the portal vein, indicating ongoing liver injury despite avacopan withdrawal. This case report highlights the hepatotoxic potential of avacopan, the importance of vigilant liver function monitoring, and the value of liver biopsy in the chronic phase of avacopan-induced liver injury. Further research is required to elucidate avacopan hepatotoxicity mechanisms and identify potential risk factors for drug-induced liver injury in patients with antineutrophil cytoplasmic antibody-associated vasculitis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"163-167"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142645345","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

Unique presentation of acute neuro-Behçet's disease involving a cytotoxic oedema core surrounded by vasogenic oedema. 急性神经性贝赫切特病的独特表现：细胞毒性水肿核心被血管源性水肿包围。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae032

Shohei Yamashita, Daiki Fujimori, Shigemoto Igari, Yusuke Yamamoto, Takahiro Mizuuchi, Hiroaki Mori, Haeru Hayashi, Koichiro Tahara, Tetsuji Sawada

{"title":"Unique presentation of acute neuro-Behçet's disease involving a cytotoxic oedema core surrounded by vasogenic oedema.","authors":"Shohei Yamashita, Daiki Fujimori, Shigemoto Igari, Yusuke Yamamoto, Takahiro Mizuuchi, Hiroaki Mori, Haeru Hayashi, Koichiro Tahara, Tetsuji Sawada","doi":"10.1093/mrcr/rxae032","DOIUrl":"10.1093/mrcr/rxae032","url":null,"abstract":"A 53-year-old woman with recurrent stomatitis, genital ulcers, and folliculitis was admitted to Tokyo Medical University Hospital after experiencing visual disturbances for the past 2 weeks and a nonthrobbing headache for the past 3 days. She had also developed numbness in her left extremities. An ophthalmological examination revealed inflammatory changes in the eye. Cerebrospinal fluid analysis showed increased cell counts, protein, and interleukin-6 levels. Brain magnetic resonance imaging revealed multiple high signal intensities on T2-weighted/fluid-attenuated inversion recovery images of the pons and occipital and parietal lobes. The T2-weighted/fluid-attenuated inversion recovery high-signal-intensity lesion in the pons was hyperintense on diffusion-weighted imaging and hypointense on apparent diffusion coefficient mapping, suggesting cytotoxic oedema. Another high-signal-intensity lesion on T2-weighted/fluid-attenuated inversion recovery was isointense to hyperintense on diffusion-weighted imaging and hyperintense on apparent diffusion coefficient, indicating vasogenic oedema. The vasogenic oedema in the left occipital lobe contained a small core that was hyperintense on diffusion-weighted imaging and hypointense on apparent diffusion coefficient, suggesting cytotoxic oedema. The patient was diagnosed with acute neuro-Behçet's disease and responded well to high-dose glucocorticoid and colchicine treatment. The present report emphasises that patients with acute neuro-Behçet's disease may present with cytotoxic oedema in the pons and cerebral spheres. Further reports of similar cases would contribute to a better understanding of the role of cytotoxic oedema in the pathophysiology of neuro-Behçet's disease and help elucidate the mechanisms underlying a unique presentation characterised by a central cytotoxic oedema core within vasogenic oedema.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"188-192"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141319400","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 Japanese case of VEXAS syndrome after COVID-19 vaccination: Comparison with previously reported cases. 日本一例接种 COVID-19 疫苗后出现的 VEXAS 综合征：与先前报告病例的比较。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae054

Yui Miyagi, Hiroshi Kobayashi, Yoshihiro Umebayashi, Akira Okimura, Munehide Nakatsugawa, Ayaka Maeda, Yohei Kirino, Akiko Aoki

引用次数: 0

Isolated central nervous system lymphomatoid granulomatosis in an older adult patient with systematic lupus erythematosus: A case report. 一名患有系统性红斑狼疮的老年患者的孤立性中枢神经系统淋巴肉芽肿：病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae062

Takayuki Kambe, Makoto Yamaguchi, Takayuki Katsuno, Hirokazu Sugiyama, Keisuke Kamiya, Kentaro Imai, Hiroshi Kinashi, Shogo Banno, Yasuhiko Ito, Takuji Ishimoto

{"title":"Isolated central nervous system lymphomatoid granulomatosis in an older adult patient with systematic lupus erythematosus: A case report.","authors":"Takayuki Kambe, Makoto Yamaguchi, Takayuki Katsuno, Hirokazu Sugiyama, Keisuke Kamiya, Kentaro Imai, Hiroshi Kinashi, Shogo Banno, Yasuhiko Ito, Takuji Ishimoto","doi":"10.1093/mrcr/rxae062","DOIUrl":"10.1093/mrcr/rxae062","url":null,"abstract":"Lymphomatoid granulomatosis (LYG) is a rare, T-cell-rich Epstein-Barr virus (EBV)-positive B-cell lymphoproliferative systemic disease. Only a few cases of LYG have been described in patients with autoimmune disorders, with only one case described in a patient with systemic lupus erythematosus (SLE). However, no cases of isolated central nervous system (CNS)-LYG have been reported in patients with autoimmune diseases. Since isolated CNS involvement is rare, its clinicopathological features remain incompletely understood. Herein, we report about an 85-year-old Japanese woman who was diagnosed with SLE 26 years ago and was stable and maintained on prednisone (5 mg/day) for 20 years. Twenty-six days before admission, she developed cognitive dysfunction. Brain magnetic resonance imaging (MRI) revealed multiple nodular lesions with ring-like enhancements seen on gadolinium-enhanced T1-weighted imaging. A brain biopsy was performed on the right frontal lobe lesion. Pathological findings revealed T-cell infiltration surrounding the blood vessels with fibrin deposition, a few multinucleated cells, and large atypical cells with prominent nucleoli. Large atypical cells positive for CD20 and EBV-encoded RNA were seen at a density of >100 cells per high-power field. Based on laboratory testing, imaging, and pathology findings, the patient was diagnosed with Grade III LYG. Treatment with tirabrutinib (480 mg once daily) was started. The patient achieved clinical response to treatment, as evidenced by improved mental status. In patients with SLE who present with multiple nodular or ring-like lesions on brain MRI, a brain biopsy with histological diagnosis is crucial for the exclusion of CNS-LYG.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"68-74"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142407345","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

Anaphylaxis and prolonged allergic reactions caused by intra-articular injection of diclofenac etalhyaluronate in knee osteoarthritis: A case report. 膝关节骨关节炎患者关节内注射双氯芬酸乙醛尿酸盐引起过敏性休克和长期过敏反应：病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae067

Tomohito Takeshige, Ryo Koyama, Yuta Hotchi, Kenji Kawai, Makiko Kohmaru, Takashi Akimoto, Junko Watanabe, Toshifumi Yae, Kenji Kido, Manabu Sugita, Kazuhisa Takahashi

{"title":"Anaphylaxis and prolonged allergic reactions caused by intra-articular injection of diclofenac etalhyaluronate in knee osteoarthritis: A case report.","authors":"Tomohito Takeshige, Ryo Koyama, Yuta Hotchi, Kenji Kawai, Makiko Kohmaru, Takashi Akimoto, Junko Watanabe, Toshifumi Yae, Kenji Kido, Manabu Sugita, Kazuhisa Takahashi","doi":"10.1093/mrcr/rxae067","DOIUrl":"10.1093/mrcr/rxae067","url":null,"abstract":"Osteoarthritis (OA) is a common condition. The treatment of knee OA aims to improve the quality of life and clinical symptoms, mainly knee pain. To develop a new treatment option, diclofenac etalhyaluronate (ONO-5704/SI-613; trade name: JOYCLU) was developed. Its anti-inflammatory and pain-relieving effects have been demonstrated. Conversely, adverse events have also been reported. Here, we report the case of a 71-year-old female who presented with anaphylaxis and prolonged allergic reactions after an intra-articular injection of JOYCLU. A basophil activation test (BAT) was performed to determine the cause of the anaphylaxis. BAT showed no positive findings. As the incidence of knee OA increases, the number of cases of JOYCLU use is expected to grow; therefore, careful administration is required. In this case, the cause was unknown by BAT. Thus, further development of appropriate testing methods is necessary. Intra-articular allergic reactions in knee OA may be modified and worsened besides normal allergic reactions; therefore, caution is required.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"53-56"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142515544","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

Arthroscopic treatment of septic arthritis of the ankle joint caused by streptococcus in patients with chronic inflammatory diseases: Two case reports. 慢性炎症患者链球菌引起的踝关节化脓性关节炎的关节镜治疗：两例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae041

Gensuke Okamura, Makoto Hirao, Takaaki Noguchi, Yuki Etani, Kosuke Ebina, Taihei Miura, Hideki Tsuboi, Atsushi Goshima, Seiji Okada, Jun Hashimoto

引用次数: 0