Modern rheumatology case reports最新文献_第6页

Successful treatment of aggressive AA amyloidosis with tocilizumab in a patient with polymyalgia rheumatica. 多发性风湿性关节炎患者使用托珠单抗成功治疗侵袭性 AA 淀粉样变性。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae050

Nuh Atas, Ali Rıza Çalışkan, Ayşenur Akatlı

引用次数: 0

Epstein-Barr virus-encoded RNA-positive lymphocytes in bone marrow and lymph nodes in an autopsy case of TAFRO syndrome. 一例 TAFRO 综合征尸检病例骨髓和淋巴结中的 Epstein-Barr 病毒编码 RNA 阳性淋巴细胞

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae049

Nao Tsugita, Ippei Miyagawa, Shingo Nakayamada, Aya Nawata, Shumpei Kosaka, Masanobu Ueno, Yoshiya Tanaka

{"title":"Epstein-Barr virus-encoded RNA-positive lymphocytes in bone marrow and lymph nodes in an autopsy case of TAFRO syndrome.","authors":"Nao Tsugita, Ippei Miyagawa, Shingo Nakayamada, Aya Nawata, Shumpei Kosaka, Masanobu Ueno, Yoshiya Tanaka","doi":"10.1093/mrcr/rxae049","DOIUrl":"10.1093/mrcr/rxae049","url":null,"abstract":"A 55-year-old man was admitted to the hospital with vomiting, diarrhoea, and chest pain. Upon examination, he exhibited signs of increased inflammatory response, acute kidney injury, and thrombocytopenia, leading to a diagnosis of TAFRO syndrome, which was supported by the clinical evidence of generalised lymphadenopathy, pleural effusion, and hepatosplenomegaly. Despite receiving intensive multimodal immunosuppressive therapy, including glucocorticoid pulse therapy (methylprednisolone 1000 mg/day), tocilizumab, and cyclosporine in the intensive care unit, the patient showed minimal response and succumbed to the disease on the seventh day of hospitalisation. Histopathological analysis of the lymph nodes revealed idiopathic multicentric Castleman disease-like features, and Epstein-Barr virus-encoded RNA (EBER) in situ hybridisation identified multiple EBER-positive cells. These findings highlight the elusive pathogenic mechanism of TAFRO syndrome and the potential resistance of some patients to standard treatments such as tocilizumab. The presence of EBER-positive cells in lymph nodes or bone marrow may serve as an indicator of disease severity and treatment resistance. Therefore, histopathological detection of EBER-positive cells may help predict responsiveness to conventional treatments, disease severity, and prognosis in patients with TAFRO syndrome.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"179-183"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142057718","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

Prolonged eosinophilia caused by Strongyloidiasis in a patient with rheumatoid arthritis treated with upadacitinib. 一名接受乌达帕替尼治疗的类风湿性关节炎患者因斯特龙线虫病引起的长期嗜酸性粒细胞增多。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae061

Marko Barešić, Paula Kilić, Mirjana Balen Topić, Branimir Anić

引用次数: 0

Pericardial artery lymphoma in a patient with dermatomyositis: A case report. 皮肌炎患者的心包动脉淋巴瘤：病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae065

Kei Hirose, Takayuki Katsuyama, Natsuki Kubota, Yuya Terajima, Kazuya Matsumoto, Kenta Shidahara, Takato Nakadoi, Shoichi Nawachi, Yu Katayama, Yoshia Miyawaki, Eri Katsuyama, Mariko Narazaki-Takano, Yoshinori Matsumoto, Noboru Asada, Ken-Ei Sada, Jun Wada

{"title":"Pericardial artery lymphoma in a patient with dermatomyositis: A case report.","authors":"Kei Hirose, Takayuki Katsuyama, Natsuki Kubota, Yuya Terajima, Kazuya Matsumoto, Kenta Shidahara, Takato Nakadoi, Shoichi Nawachi, Yu Katayama, Yoshia Miyawaki, Eri Katsuyama, Mariko Narazaki-Takano, Yoshinori Matsumoto, Noboru Asada, Ken-Ei Sada, Jun Wada","doi":"10.1093/mrcr/rxae065","DOIUrl":"10.1093/mrcr/rxae065","url":null,"abstract":"A woman in her 60s presented with erythema on both elbows, dyspnea on exertion, discomfort, and swelling of the left upper extremity, followed by swelling and myalgia of the right upper and lower extremities. She was diagnosed with anti-transcriptional intermediary factor 1-γ antibody-positive dermatomyositis with interstitial pneumonia. The initial screening tests for malignant diseases including contrast-enhanced computed tomography, upper and lower endoscopy, and gynecological examination did not reveal any obvious abnormalities. The patient experienced two recurrent episodes of muscle weakness and dysphagia during treatment with intravenous glucocorticoids and cyclophosphamide. Five months after diagnosis, a bone marrow biopsy and positron emission tomography-computed tomography scan revealed a coronary malignant lymphoma with suspected systemic metastasis. Although chemotherapy was initiated, the patient ultimately succumbed to alveolar haemorrhage. Coronary lymphoma is very rare and there has been no report of cases associated with myositis. Positron emission tomography-computed tomography may be useful for searching malignancy in anti-transcriptional intermediary factor 1-γ antibody-positive dermatomyositis cases of recurrent relapse.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"100-103"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142712340","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

Jaccoud's Arthropathy Secondary to Leprosy. 继发于麻风病的贾科德关节病。

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Modern rheumatology case reports Pub Date : 2025-01-09 DOI: 10.1093/mrcr/rxae077

Izabela Prado Viana, Maria de Lourdes Castro de Oliveira Figueiroa, Beatriz Paixão Argollo, Gustavo Luiz Behrens Pinto, Mittermayer Barreto Santiago

引用次数: 0

Constrictive pericarditis as first presentation of IgG4-related disease: pitfalls and pearls. 缩窄性心包炎是igg4相关疾病的首次表现：陷阱和珍珠

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Modern rheumatology case reports Pub Date : 2024-12-26 DOI: 10.1093/mrcr/rxae089

Timothy N Kwan, Gemma Kwan, John Yiannikas

引用次数: 0

MPO-ANCA-positive eosinophilic granulomatosis with polyangiitis complicated by alveolar hemorrhage treated with mepolizumab as an induction therapy: Case report. mpo - anca阳性嗜酸性粒细胞肉芽肿病合并多血管炎合并肺泡出血，mepolizumab作为诱导治疗：病例报告。

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Modern rheumatology case reports Pub Date : 2024-12-21 DOI: 10.1093/mrcr/rxae088

Mana Yoshida, Shigeru Iwata, Kayoko Tabata, Aya Hashimoto, Ryo Matsumiya, Katsunori Tanaka, Ryuta Iwamoto, Masatoshi Jinnin, Takao Fujii

{"title":"MPO-ANCA-positive eosinophilic granulomatosis with polyangiitis complicated by alveolar hemorrhage treated with mepolizumab as an induction therapy: Case report.","authors":"Mana Yoshida, Shigeru Iwata, Kayoko Tabata, Aya Hashimoto, Ryo Matsumiya, Katsunori Tanaka, Ryuta Iwamoto, Masatoshi Jinnin, Takao Fujii","doi":"10.1093/mrcr/rxae088","DOIUrl":"https://doi.org/10.1093/mrcr/rxae088","url":null,"abstract":"Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic vasculitis preceded by bronchial asthma or allergic sinusitis and accompanied by peripheral blood eosinophilia. Immunosuppressive drugs, such as cyclophosphamide in addition to high-dose glucocorticoids, are recommended for induction of remission in patients with severe EGPA. Although mepolizumab is widely recognized as remission induction therapy in non-fatal/non-organ disabling or relapsed/refractory EGPA, its efficacy and safety in induction of remission for severe cases have been ambiguous. In this context, we report a case of MPO-ANCA-positive severe EGPA in which the patient had a favorable course using mepolizumab as an induction remission therapy. The patient, a 74-year-old man, had MPO-ANCA-positive severe EGPA with alveolar hemorrhage. High-dose glucocorticoids and intravenous cyclophosphamide were started as remission induction therapy. However, after the initiation of intravenous cyclophosphamide, alveolar hemorrhage worsened, and there was development of opportunistic infections, such as aspergillus and cytomegalovirus antigenemia. Treatment with the antifungal drug voriconazole and the antiviral drug ganciclovir was started for opportunistic infection, and the treatment for EGPA was switched from intravenous cyclophosphamide to mepolizumab. As a result, alveolar hemorrhage improved, glucocorticoids were reduced, and the infection also improved. Mepolizumab as remission induction therapy for severe EGPA were thought to be appropriate and effective treatment in this case. However, the efficacy and safety of mepolizumab for this purpose require comprehensive evaluation.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142873758","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 glucocorticoid-refractory IgA vasculitis with diffuse alveolar hemorrhage: a therapeutic strategy for aberrant immunoglobulin depletion. 糖皮质激素难治性IgA血管炎伴弥漫性肺泡出血一例：异常免疫球蛋白耗竭的治疗策略。

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Modern rheumatology case reports Pub Date : 2024-12-19 DOI: 10.1093/mrcr/rxae087

Kazuhiko Hirokawa, Shunichi Sato, Eiji Hiraoka, Keiichi Iwanami

{"title":"A case of glucocorticoid-refractory IgA vasculitis with diffuse alveolar hemorrhage: a therapeutic strategy for aberrant immunoglobulin depletion.","authors":"Kazuhiko Hirokawa, Shunichi Sato, Eiji Hiraoka, Keiichi Iwanami","doi":"10.1093/mrcr/rxae087","DOIUrl":"https://doi.org/10.1093/mrcr/rxae087","url":null,"abstract":"Diffuse alveolar hemorrhage (DAH) is a rare and severe complication of IgA vasculitis (IgAV). Although glucocorticoids and immunosuppressive agents are used for its treatment, there is no consensus on the optimal form of treatment. We herein report the case of a 53-year-old, female patient with IgAV. She was initially resistant to glucocorticoid therapy and experienced acute respiratory failure due to DAH but responded well to rituximab (RTX) and plasma exchange (PLEX). While some previous case reports have suggested that RTX or PLEX can be effective for severe IgAV, there are no reports of a combination of RTX and PLEX being used successfully to treat IgAV-associated DAH. In the model of IgAV pathogenesis proposed herein, aberrant IgA1 and IgA-specific IgG autoantibodies play a pivotal role. PLEX may facilitate the prompt removal of these circulating, aberrant immunoglobulins while RTX inhibits their further production. Consequently, a combination of RTX and PLEX may represent an effective treatment approach for severe glucocorticoid-refractory cases of IgAV.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142857419","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 Erdheim-Chester disease-a mimicker of IgG4-related disease and large vessel vasculitis. 一例埃尔德海姆-切斯特病--IgG4 相关疾病和大血管炎的模拟者。

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Modern rheumatology case reports Pub Date : 2024-12-17 DOI: 10.1093/mrcr/rxae086

Ippei Miyagawa, Shingo Nakayamada, Hirotsugu Nohara, Shumpei Kosaka, Masanobu Ueno, Yoshiya Tanaka

{"title":"A case of Erdheim-Chester disease-a mimicker of IgG4-related disease and large vessel vasculitis.","authors":"Ippei Miyagawa, Shingo Nakayamada, Hirotsugu Nohara, Shumpei Kosaka, Masanobu Ueno, Yoshiya Tanaka","doi":"10.1093/mrcr/rxae086","DOIUrl":"https://doi.org/10.1093/mrcr/rxae086","url":null,"abstract":"The patient was a 57-year-old man who developed bilateral thigh pain and chest tightness one year ago. Chest CT scan showed reticular shadows, thickened interlobular septa in both lung fields, and pericardial effusion. Three months ago, his symptoms worsened. A contrast CT scan revealed increased pericardial effusion, multiple masses in the right atrium, soft tissue shadows suggestive of retroperitoneal fibrosis, and soft tissue shadows around the thoracic and abdominal aorta. He visited our hospital, suspecting IgG4-related disease (IgG4-RD) or large vessel vasculitis (LVV). Based on the involvement of various organs and bilateral thigh pain, Erdheim-Chester disease (ECD) was suspected, and an FDG-PET scan was performed. In addition to increased accumulation around the right ventricle, right coronary artery, and aorta, increased accumulation was confirmed in the distal femurs and proximal tibias on both sides, strongly suggesting ECD. A bone biopsy confirmed the diagnosis of ECD, showing bone fibrosis with CD68-positive and CD1a-negative foam cell infiltration, which is a characteristic of ECD. ECD is an extremely rare form of non-Langerhans cell histiocytosis. ECD affects a wide variety of organs, and its imaging findings can sometimes resemble those of IgG4-related disease or LVV. However, bone lesions are characteristic of ECD and are a key finding for its diagnosis. When systemic organ lesions, including bone lesions, are present, ECD should be included in the differential diagnosis, and PET-CT should be considered.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142839753","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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Utility of external fixation for traumatic periprosthetic fracture after total ankle arthroplasty in patients with rheumatoid arthritis: A report of two cases. 类风湿性关节炎患者全踝关节置换术后创伤性假体周围骨折外固定的实用性：两个病例的报告。

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Modern rheumatology case reports Pub Date : 2024-12-14 DOI: 10.1093/mrcr/rxae084

Gensuke Okamura, Takaaki Noguchi, Yuki Etani, Kosuke Ebina, Seiji Okada, Jun Hashimoto, Makoto Hirao

引用次数: 0