Modern rheumatology case reports最新文献_第4页

Paradoxical pyoderma gangrenosum secondary to adalimumab successfully treated with methotrexate: A case report. 甲氨蝶呤成功治疗阿达木单抗继发的矛盾性坏疽性脓皮病1例。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxae073

Kristy El Morr, Mohamad Ali Rida

引用次数: 0

A case of polyarteritis nodosa with severe lower limb ulcer that was treated with prednisolone and tocilizumab. 强的松龙联合托珠单抗治疗结节性多动脉炎合并严重下肢溃疡1例。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxae085

Naoto Okubo, Yuki Oba, Daisuke Ikuma, Hiroki Mizuno, Masayuki Yamanouchi, Tatsuya Suwabe, Yoshifumi Ubara, Naoki Sawa

{"title":"A case of polyarteritis nodosa with severe lower limb ulcer that was treated with prednisolone and tocilizumab.","authors":"Naoto Okubo, Yuki Oba, Daisuke Ikuma, Hiroki Mizuno, Masayuki Yamanouchi, Tatsuya Suwabe, Yoshifumi Ubara, Naoki Sawa","doi":"10.1093/mrcr/rxae085","DOIUrl":"10.1093/mrcr/rxae085","url":null,"abstract":"Polyarteritis nodosa (PAN) is a rare systemic necrotising vasculitis that can lead to the formation of refractory lower limb ulcers requiring amputation. The standard treatment for severe PAN involves combination therapy with steroids and cyclophosphamide; however, some cases prove to be challenging. Recently, case reports have described the use of biological agents for PAN treatment. We present the case of a 61-year-old Japanese man with cutaneous PAN and refractory recurrent lower limb ulcers. In 2017, the patient was admitted to the hospital because of exacerbation of a right lower limb ulcer. Despite combination therapy with corticosteroids, cyclophosphamide, and endovascular therapy, the gangrene in the right lower leg progressed, and amputation was performed. The patient was temporarily stabilised with prednisolone monotherapy. In 2019, new ulcers were observed on the left lower limb. Owing to steroid resistance, subcutaneous tocilizumab (162 mg/week) was initiated. Over a few months, the ulcer healed completely, and left lower limb amputation was avoided. Therefore, tocilizumab could potentially be one of the treatment options for severe cases in the future.","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":"142824935","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 autopsy of a patient with polyarteritis nodosa who developed fatal intestinal perforation accompanied by systemic multiple aneurysms and arterial thrombosis. 一例结节性多动脉炎并发致命性肠穿孔并伴有全身多发动脉瘤和动脉血栓形成的尸检报告。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxae079

Nao Tsugita, Ippei Miyagawa, Masanobu Ueno, Miyabi Takahashi, Shumpei Kosaka, Shingo Nakayamada, Yoshiya Tanaka

{"title":"An autopsy of a patient with polyarteritis nodosa who developed fatal intestinal perforation accompanied by systemic multiple aneurysms and arterial thrombosis.","authors":"Nao Tsugita, Ippei Miyagawa, Masanobu Ueno, Miyabi Takahashi, Shumpei Kosaka, Shingo Nakayamada, Yoshiya Tanaka","doi":"10.1093/mrcr/rxae079","DOIUrl":"10.1093/mrcr/rxae079","url":null,"abstract":"The patient was a 48-year-old man who had developed acute myocardial infarction 3 years earlier. He started experiencing recurrent attacks of abdominal pain 2 years earlier. One month before the presentation, he developed perforative peritonitis, which was treated with right hemicolectomy. Preoperative computed tomography revealed systemic thrombotic aneurysms and fibrinoid necrotising vasculitis was detected in the vessels of the serosa of the resected intestinal specimen. These findings led to a diagnosis of polyarteritis nodosa. Despite the start of remission induction therapy with high-dose glucocorticoid and intermittent intravenous cyclophosphamide, the effect of immunosuppressive therapy was limited. Approximately 1 month after treatment initiation, he died from small intestinal perforation. Polyarteritis nodosa often exhibits nonspecific clinical symptoms, which make an early diagnosis difficult in some cases. Although the prognosis depends on the presence of ischaemic lesions due to a ruptured aneurysm or intra-aneurysmal thrombi, it is not rare for the diagnosis to be made following acute myocardial infarction or acute abdominal pain. In young patients with iscahemic organ dysfunction without any arteriosclerotic lesions at low risk of developing cardiovascular events, early diagnosis can be made by performing a whole-body examination with a differential diagnosis of polyarteritis nodosa.","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":"142808998","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

Correction to: A case of atypical IgG4-related disease presenting hypereosinophilia, polyneuropathy, and liver dysfunction. 更正:1例非典型igg4相关疾病，表现为嗜酸性粒细胞增多、多发性神经病变和肝功能障碍。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxad066

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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 : 2025-07-25 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":"10.1093/mrcr/rxae086","url":null,"abstract":"The patient was a 57-year-old man who developed bilateral thigh pain and chest tightness 1 year ago. Chest computed tomography (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 University Hospital of University of Occupational and Environmental Health, Japan 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 18F-fluorodeoxyglucose Positron Emission Tomography (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":"2025-07-25","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}

引用次数: 0

A case of glucocorticoid-refractory IgA vasculitis with diffuse alveolar haemorrhage: 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 : 2025-07-25 DOI: 10.1093/mrcr/rxae087

Kazuhiko Hirokawa, Shunichi Sato, Eiji Hiraoka, Keiichi Iwanami

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Above-knee amputation in a 69-year-old male with gouty tophi complicated by necrotising fasciitis - a case report. 69岁男性痛风性痛风伴坏死性筋膜炎行膝上截肢1例。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxae080

Metasebia Worku Abebe, Abigya Aschalew Asfaw

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Unusual systemic conditions in a patient with giant cell arteritis. 巨细胞动脉炎患者的异常全身状况。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf014

Khalid El-Jack, Fawaz Naeem, Jenelle Safadi, Dario Marangoni, Alexander M Solomon, Franz Fogt, Grant T Liu, Madhura A Tamhankar

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Effect of abatacept added to mycophenolate mofetil for refractory calcinosis in juvenile dermatomyositis. 加入霉酚酸酯治疗青少年皮肌炎难治性钙化的效果。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf004

Maho Shimizu, Haruna Nakaseko, Yoshinao Muro, Naomi Iwata

{"title":"Effect of abatacept added to mycophenolate mofetil for refractory calcinosis in juvenile dermatomyositis.","authors":"Maho Shimizu, Haruna Nakaseko, Yoshinao Muro, Naomi Iwata","doi":"10.1093/mrcr/rxaf004","DOIUrl":"10.1093/mrcr/rxaf004","url":null,"abstract":"Calcinosis is an intractable condition in juvenile dermatomyositis (JDM). The effect of abatacept on calcinosis remains controversial. We describe a case of an 8-year-old boy in whom the addition of abatacept to mycophenolate mofetil was effective against calcinosis in JDM. The patient with antinuclear matrix protein 2 autoantibody suffered from refractory jJDM despite long-term treatment with corticosteroids and various immunosuppressive agents. Subcutaneous calcinosis with repeated spontaneous pain at the calcinosis site emerged 2 years after the start of treatment and gradually increased despite clinical improvement in muscle symptoms. The addition of intravenous abatacept to mycophenolate mofetil at the age of 6 years halted the increase in calcinosis. Spontaneous pain in calcinosis disappeared within 2 months. The calcinosis had significantly decreased by 1 year. An important strategy on calcinosis in JDM is getting complete control of the inflammation. Our case report suggests that abatacept may contribute to the improvement of calcinosis in JDM, at least as part of combination therapy with mycophenolate mofetil.","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":"143607500","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

Neurosarcoidosis or granulomatosis with polyangiitis? A complex case of a brain mass. 神经结节病还是肉芽肿伴多血管炎？一个复杂的脑肿块病例。

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Modern rheumatology case reports Pub Date : 2025-07-25 DOI: 10.1093/mrcr/rxaf019

Luisa Fernanda Jiménez-Arcia, Estiven Crespo-Vizcaíno, Alexandra González-Montoya, Alejandro Vélez-Hoyos, Luis Fernando Pinto-Peñaranda

{"title":"Neurosarcoidosis or granulomatosis with polyangiitis? A complex case of a brain mass.","authors":"Luisa Fernanda Jiménez-Arcia, Estiven Crespo-Vizcaíno, Alexandra González-Montoya, Alejandro Vélez-Hoyos, Luis Fernando Pinto-Peñaranda","doi":"10.1093/mrcr/rxaf019","DOIUrl":"10.1093/mrcr/rxaf019","url":null,"abstract":"Sarcoidosis is an immune-mediated systemic disease characterised by the presence of non-caseating granulomas in various parts of the body in the absence of another defined aetiology. Neurologic involvement [neurosarcoidosis (NS)], which occurs in 5-10% of patients with the disease, encompasses a range of clinical and histopathological manifestations that can lead to significant morbidity and mortality. We present a case of a young man with a history of chronic sinusitis, who developed sudden headache associated with seizures. After thorough clinical and paraclinical evaluation, the diagnosis of NS was made once other neurovascular, infectious, metabolic, tumour-related, and immune-mediated aetiologies were ruled out. NS can present as a large dural mass due to nodular pachymeningitis, which can be clinically indistinguishable from other entities such as neoplasms and granulomatosis with polyangiitis. Isolated central nervous system involvement in this entity is rare and usually it is associated with other systemic manifestations. More aggressive management is required to treat this form of sarcoidosis presentation. NS represents a diagnostic challenge and requires ruling out more common entities such as infectious and non-infectious causes like granulomatosis with polyangiitis.","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":"143652905","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