Modern rheumatology case reports最新文献_第5页

Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities. 伴有口腔和喉部受累的多中心网状组织细胞增多症与佐剂诱发的自身免疫/炎症综合征（ASIA）：扩展两种不常见实体的谱系。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae040

Ilaria Girolami, Reinhard Walter Kluge, Lorenz Gallmetzer, Cecilia Albi, Paola Sorgi, Christian Dejaco, Martina Tauber, Esther Hanspeter, Peter Matzneller

{"title":"Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities.","authors":"Ilaria Girolami, Reinhard Walter Kluge, Lorenz Gallmetzer, Cecilia Albi, Paola Sorgi, Christian Dejaco, Martina Tauber, Esther Hanspeter, Peter Matzneller","doi":"10.1093/mrcr/rxae040","DOIUrl":"10.1093/mrcr/rxae040","url":null,"abstract":"Multicentric reticulohistiocytosis (MRH) is the most frequent entity in the group of reticulohistiocytoses. It is usually accompanied by symmetrical erosive polyarthritis and is frequently associated with cancer and autoimmune disorders. Autoimmune/inflammatory syndrome induced by adjuvants (ASIA) is an inflammatory syndrome triggered by adjuvants such as those contained in vaccines or by silicone implants. Here, we report the case of a 71-year-old woman with a history of breast cancer treated with surgery and subsequent prosthesis who developed a systemic hyperinflammatory syndrome including seronegative symmetric polyarthritis, multiple skin lesions, and two large nodular lesions in the oral cavity and larynx. The clinical picture was consistent with a clinical diagnosis of ASIA, with breast implant rupture and/or vaccination against severe acute respiratory syndrome Coronavirus 2 (SARS-CoV-2) as possible triggers. Histopathology of skin, oral nodules, and laryngeal nodules revealed cutaneous/mucous and submucosal infiltration of large epithelioid mononuclear or binucleated cells with fine granular ground glass-like cytoplasm and round to kidney-shaped nuclei with prominent nucleoli, without atypical features or relevant pleomorphism, accompanied by sparse giant cells and lymphocytes. These cells stained positive for CD68 and CD45 and negative for S100, CD1a, and markers of epithelial or neural/melanocytic differentiation, altogether consistent with a diagnosis of reticulohistiocytosis. Clinicopathological correlation allowed the final diagnosis of MRH. To our knowledge, this is the first report of a co-occurrence of MRH with ASIA, and this is relevant to broaden the spectrum of both these rare diseases.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"200-206"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141891405","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

Kikuchi-Fujimoto disease and primary Sjögren's syndrome coexisting: A case-based literature review. 菊池-藤本氏病与原发性斯约格伦综合征并存：基于病例的文献综述。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae058

Maria João Cadório, João Oliveira, João Gama, Cátia Duarte

{"title":"Kikuchi-Fujimoto disease and primary Sjögren's syndrome coexisting: A case-based literature review.","authors":"Maria João Cadório, João Oliveira, João Gama, Cátia Duarte","doi":"10.1093/mrcr/rxae058","DOIUrl":"10.1093/mrcr/rxae058","url":null,"abstract":"Kikuchi-Fujimoto Disease (KFD), also known as Kikuchi histiocytic necrotising lymphadenitis, is an extremely rare and benign condition that mostly affects young women. It is characterised by lymph node involvement with a predilection for the cervical region, commonly presenting with tender lymphadenopathy and a low-grade fever. The diagnosis requires excisional lymph node biopsy with immunohistochemical analysis. KFD is mostly self-limiting within a few weeks to months, with only some patients requiring symptomatic relief with non-steroidal anti-inflammatory drugs or corticosteroids, and a minority developing recurrent episodes of the disease. Importantly, it has been reported in association with systemic lupus erythematosus and, to a lesser extent, other immune-mediated inflammatory rheumatic diseases, such as Sjögren's syndrome, whose clinical presentation itself may include lymphadenopathy. In this paper, we present an unusual case of a woman with primary Sjögren syndrome (pSS) and a past medical history relevant for lymphoma, sarcoidosis, and thymoma, who later developed KFD, a particularly challenging diagnosis in this setting. We then performed a literature review of the association between KFD and pSS, gathering a total of 13 patients, and focusing epidemiological, clinical, and laboratory data.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"110-116"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142305106","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 Buerger's disease with vasculopathy and skin fibrosis requiring differential diagnosis from systemic sclerosis. 一例布格氏病伴有血管病变和皮肤纤维化，需要与系统性硬化症进行鉴别诊断。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae036

Masashi Funada, Shingo Nakayamada, Shunsuke Fukuyo, Satoshi Kubo, Aya Nawata, Yuya Fujita, Yoshiya Tanaka

{"title":"A case of Buerger's disease with vasculopathy and skin fibrosis requiring differential diagnosis from systemic sclerosis.","authors":"Masashi Funada, Shingo Nakayamada, Shunsuke Fukuyo, Satoshi Kubo, Aya Nawata, Yuya Fujita, Yoshiya Tanaka","doi":"10.1093/mrcr/rxae036","DOIUrl":"10.1093/mrcr/rxae036","url":null,"abstract":"Buerger's disease is characterised by peripheral ischaemia due to occlusion of small- and medium-sized arteries in the extremities. This report describes a case of Buerger's disease in a 51-year-old male who presented with findings resembling systemic sclerosis (SSc). The patient exhibited Raynaud's phenomenon in year 2015, which developed to skin hardening, nail avulsion, and ulceration of the right fingers in year 2018. Diagnostic testing showed positive microvasculopathy on nailfold videocapillaroscopy (NVC) and positive fibrosis on skin biopsy. Although the patient fulfilled the 2013 American College of Rheumatology/European League Against Rheumatism classification criteria for SSc, several findings in this case were atypical for SSc, including left-right asymmetry in finger involvement, nail loss, and negative autoantibody tests. Contrast-enhanced computed tomography showed poor perfusion of the right ulnar artery, and a heavy smoking history was established in the patient case. Therefore, based on Shionoya's criteria, he was diagnosed with a case of Buerger's disease confined to the upper extremity. Smoking cessation and vasodilator therapy resulted in the prompt resolution of ischaemic symptoms, skin hardening, and ulcerations. Furthermore, NVC abnormalities improved, and ulnar artery occlusion showed reperfusion on repeat testing. The present case suggests that hypoxaemia-driven microvasculopathy may contribute to vascular occlusion and skin fibrosis observed in this atypical presentation.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"127-130"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141857474","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

Elderly-onset inflammatory myopathy associated with Sjögren's syndrome following SARS-CoV-2 vaccination. 接种 SARS-CoV-2 疫苗后，老年炎症性肌病与斯约格伦综合征相关。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae055

Yuta Komori, Satomi Kobayashi, Keiko Hatano, Yuko Saito, Tomio Arai, Kanae Kubo

{"title":"Elderly-onset inflammatory myopathy associated with Sjögren's syndrome following SARS-CoV-2 vaccination.","authors":"Yuta Komori, Satomi Kobayashi, Keiko Hatano, Yuko Saito, Tomio Arai, Kanae Kubo","doi":"10.1093/mrcr/rxae055","DOIUrl":"10.1093/mrcr/rxae055","url":null,"abstract":"As vaccination against SARS-CoV-2 has progressed, various autoimmune diseases, including inflammatory myopathies, have been reported to develop after vaccination. Sjögren's syndrome (SS) sometimes presents as extra-glandular manifestations including inflammatory myopathy. In this report, we describe a case of inflammatory myopathy associated with SS that occurred in an atypically elderly patient after receiving the first dose of the SARS-CoV-2 mRNA vaccine (BNT162b2). The inflammatory myopathy was pathologically classified into non-specific myositis and characterised by predominant infiltration of the B cell lineage in this case. Combined treatment with glucocorticoid, intravenous immunoglobulin, and immunosuppressant resulted in an improvement in swallowing function and muscle strength. While we recognise the efficacy and safety of SARS-CoV-2 vaccines, we also emphasise the importance of recognising that individuals with an immunogenetic predisposition such as positivity of anti SS-A antibody may show disease activity including inflammatory myopathy following vaccination in SS, even at an atypically old age.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"104-109"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142305105","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

Symptomatic pachydermodactyly: A case report. 有症状的畸形：病例报告

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae064

Ayaka Ito, Hirotaka Tsuno, Yusuke Yano, Shinichi Nogi, Yoshihiko Tabira, Hanako Ohmatsu, Shogo Honda, Ayako Horita, Ikuo Saito, Toshihiro Matsui

{"title":"Symptomatic pachydermodactyly: A case report.","authors":"Ayaka Ito, Hirotaka Tsuno, Yusuke Yano, Shinichi Nogi, Yoshihiko Tabira, Hanako Ohmatsu, Shogo Honda, Ayako Horita, Ikuo Saito, Toshihiro Matsui","doi":"10.1093/mrcr/rxae064","DOIUrl":"10.1093/mrcr/rxae064","url":null,"abstract":"Pachydermodactyly, an uncommon variant of digital fibromatosis, predominantly affects young men and results in fibrous swelling on the sides of the proximal interphalangeal joints. It is generally considered benign and asymptomatic, requiring only regular follow-up. Nevertheless, the absence of established diagnostic criteria has led to misdiagnoses, prompting the administration of unnecessary medications. In this report, we present a 14-year-old Japanese male with symptomatic pachydermodactyly (PDD), necessitating careful differentiation from juvenile idiopathic arthritis (JIA) due to the presence of morning stiffness. Despite exhibiting typical PDD features, the patient's age and symptoms suggested rheumatoid factor-negative polyarticular JIA. However, the lack of inflammatory findings precluded a JIA diagnosis. Following confirmation of the absence of uveitis and progression of bone destruction, the morning stiffness spontaneously resolved without active treatment. However, the patient underwent surgery for aesthetic reasons to alleviate the persistent swelling. Our case highlights the nuances of symptomatic PDD, with a literature review revealing similarities between symptomatic and asymptomatic cases. This challenges the suitability of asymptomatic status as a definitive diagnostic criterion. Our findings contribute to the ongoing efforts in establishing diagnostic criteria for PDD, aiming to reduce misdiagnoses and unnecessary medications in patients.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"224-227"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142524007","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

New onset of giant cell arteritis with ischaemic optic neuropathy following the seventh dose of COVID-19 mRNA vaccination: A case report and literature review. 第七剂 COVID-19 mRNA 疫苗接种后新发巨细胞动脉炎伴缺血性视神经病变：病例报告和文献综述。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae042

Shin-Ichiro Ohmura, Haruka Yonezawa, Toshitaka Yukishima, Yuko Gohto, Akira Obana

{"title":"New onset of giant cell arteritis with ischaemic optic neuropathy following the seventh dose of COVID-19 mRNA vaccination: A case report and literature review.","authors":"Shin-Ichiro Ohmura, Haruka Yonezawa, Toshitaka Yukishima, Yuko Gohto, Akira Obana","doi":"10.1093/mrcr/rxae042","DOIUrl":"10.1093/mrcr/rxae042","url":null,"abstract":"Coronavirus disease (COVID-19) vaccines have demonstrated excellent efficacy in reducing the morbidity and severity of the disease. However, some patients have been reported to develop systemic rheumatic diseases, such as rheumatoid arthritis, myocarditis, Guillain-Barre syndrome, and giant cell arteritis (GCA) following COVID-19 vaccination. We present a case of GCA with ischaemic optic neuropathy following COVID-19 mRNA vaccination. A 73-year-old woman developed headache, myalgia, scalp tenderness, and jaw claudication 4 days after her seventh dose of the vaccination; she also developed severe visual disturbances 1 month after the vaccination. The blood examination tests showed an increased serum C-reactive protein level and erythrocyte sedimentation rate. The echogram for the temporal artery showed a halo sign. Ophthalmic examination revealed ischaemic optic neuropathy in both eyes. The patient was treated with a high-dose glucocorticoid and tocilizumab under the diagnosis of GCA with ischaemic optic neuropathy, obtaining mild improvement of the symptoms. This report underscores the need for clinical vigilance and further data collection regarding GCA cases after COVID-19 vaccination.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"131-136"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141891406","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

Calcium pyrophosphate crystal arthritis associated with immune checkpoint inhibitor successfully treated with intra-articular glucocorticoid: A case report. 使用关节内糖皮质激素成功治疗与免疫检查点抑制剂相关的焦磷酸钙结晶关节炎：病例报告。

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Modern rheumatology case reports Pub Date : 2025-01-16 DOI: 10.1093/mrcr/rxae029

Kazuya Abe, Taro Iwamoto, Kei Ikeda, Takahiro Sugiyama, Shunsuke Furuta, Go Saito, Hanae Wakabayashi, Hiroshi Nakajima

{"title":"Calcium pyrophosphate crystal arthritis associated with immune checkpoint inhibitor successfully treated with intra-articular glucocorticoid: A case report.","authors":"Kazuya Abe, Taro Iwamoto, Kei Ikeda, Takahiro Sugiyama, Shunsuke Furuta, Go Saito, Hanae Wakabayashi, Hiroshi Nakajima","doi":"10.1093/mrcr/rxae029","DOIUrl":"10.1093/mrcr/rxae029","url":null,"abstract":"Immune checkpoint inhibitors (ICIs) sometimes induce immune-related adverse events (irAEs), and arthritis is one of the irAE symptoms. Recently, crystal-induced arthritis, such as calcium pyrophosphate (CPP) crystal deposition disease and gout, has been reported to occur after ICI administration. However, the distinction between ICI-associated crystal arthritis and ICI-induced non-crystal arthritis is difficult because their symptoms are similar. Besides, optimal treatment for ICI-associated crystal arthritis has not been established. Here, we report a patient who developed CPP crystal arthritis twice after pembrolizumab (ICI) administration and was successfully treated with intra-articular glucocorticoid injection. He suffered arthritis and acute interstitial nephritis simultaneously after ICI administration. Musculoskeletal ultrasound of his affected joint suggests that his arthritis was crystal-induced arthritis, and arthrocentesis detected CPP crystal in synovial fluid. Thus, we diagnosed his arthritis as ICI-associated crystal arthritis. Therefore, our case encourages the use of musculoskeletal ultrasound in patients with arthritis after treatment with ICI because it may distinguish between ICI-associated crystal arthritis and ICI-induced non-crystal arthritis. Besides, ICI-associated crystal arthritis could be treatable by intra-articular glucocorticoid injection.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"27-30"},"PeriodicalIF":0.9,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140946720","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 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