Modern rheumatology case reports最新文献

MPO-ANCA-positive eosinophilic granulomatosis with polyangiitis complicated by alveolar hemorrhage treated with mepolizumab as an induction therapy: Case report.

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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.

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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。

IF 0.9

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}

引用次数: 0

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。

IF 0.9

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

A case of polyarteritis nodosa with severe lower limb ulcer that was treated with prednisolone and tocilizumab.

IF 0.9

Modern rheumatology case reports Pub Date : 2024-12-14 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":"https://doi.org/10.1093/mrcr/rxae085","url":null,"abstract":"Polyarteritis nodosa (PAN) is a rare systemic necrotizing 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 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 stabilized with PSL 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":"2024-12-14","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

IF 0.9

Modern rheumatology case reports Pub Date : 2024-12-13 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":"https://doi.org/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":"2024-12-13","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

A case report of a 14-year-old male patient with large vessel vasculitis following COVID-19.

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Modern rheumatology case reports Pub Date : 2024-12-12 DOI: 10.1093/mrcr/rxae081

Hiroki Nemoto, Yoshihiro Nozaki, Takashi Matsumoto, Kaori Kiyoki, Takumi Ishiodori, Atsushi Morita, Kazuo Imagawa, Takashi Murakami, Miho Takahashi, Hironori Imai, Hidetoshi Takada

{"title":"A case report of a 14-year-old male patient with large vessel vasculitis following COVID-19.","authors":"Hiroki Nemoto, Yoshihiro Nozaki, Takashi Matsumoto, Kaori Kiyoki, Takumi Ishiodori, Atsushi Morita, Kazuo Imagawa, Takashi Murakami, Miho Takahashi, Hironori Imai, Hidetoshi Takada","doi":"10.1093/mrcr/rxae081","DOIUrl":"https://doi.org/10.1093/mrcr/rxae081","url":null,"abstract":"Most reported cases of large vessel vasculitis (LVV) following coronavirus disease 2019 (COVID-19) have involved adults, with paediatric cases being rare. We present the case of a 14-year-old boy who developed LVV following COVID-19. Initially, he presented with fever and cough, and nasopharyngeal polymerase chain reaction testing confirmed COVID-19. His symptoms spontaneously resolved without specific COVID-19 treatments. However, 10 days after contracting COVID-19, his fever recurred, and his inflammatory markers were significantly elevated. His condition did not meet the criteria for Kawasaki disease or multisystem inflammatory syndrome in children associated with COVID-19. Contrast-enhanced computed tomography revealed arterial wall thickening in the aorta and carotid arteries, indicative of LVV. Upon initiation of high-dose immunoglobulin therapy and aspirin, his fever subsided, and his inflammatory markers and imaging findings normalised. Differential diagnosis ruled out infections, immune disorders, and Takayasu arteritis (TAK), a common cause of aortitis in children. Over a 1-year follow-up period, there was no recurrence and no stenotic lesions in large vessels. This finding suggests that the patient experienced transient LVV following COVID-19. Cytokine profile analysis performed before and after treatment revealed elevated levels of interleukin (IL)-6, IL-8, and IL-12/IL-23p40, typically associated with the active phase of TAK. Importantly, IL-17A and tumour necrosis factor-α levels were normal, as elevations in these cytokines have been linked to TAK recurrence. Notably, some cases of LVV following COVID-19 do not respond well to treatment; further research, including case accumulation and cytokine profile analysis, is needed to better predict prognosis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815435","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

Effective Use of Ultra-High Molecular Weight Polyethylene Cable and Krackow Suture for Stretched Out Patellar Tendon due to Scarring in a Case with Rheumatoid Arthritis Post-Total Knee Arthroplasty.

IF 0.9

Modern rheumatology case reports Pub Date : 2024-12-12 DOI: 10.1093/mrcr/rxae074

Eiji Kinoshita, Naoki Kondo, Osamu Tanifuji, Rika Kakutani, Nariaki Hao, Hiroyuki Kawashima

{"title":"Effective Use of Ultra-High Molecular Weight Polyethylene Cable and Krackow Suture for Stretched Out Patellar Tendon due to Scarring in a Case with Rheumatoid Arthritis Post-Total Knee Arthroplasty.","authors":"Eiji Kinoshita, Naoki Kondo, Osamu Tanifuji, Rika Kakutani, Nariaki Hao, Hiroyuki Kawashima","doi":"10.1093/mrcr/rxae074","DOIUrl":"https://doi.org/10.1093/mrcr/rxae074","url":null,"abstract":"Patellar tendon rupture is a severe complication following total knee arthroplasty. We encountered a case of rheumatoid arthritis with an incomplete rupture of the patellar tendon post-total knee arthroplasty. An 84-year-old woman was diagnosed with an incomplete rupture of the right patellar tendon 3 months post-total knee arthroplasty of her right knee. The patient exhibited a 45° extension lag 6 months post-total knee arthroplasty, necessitating reconstruction surgery. Intraoperative findings revealed incomplete rupture of the patellar tendon that was stretched out, diagnosed as incomplete patellar tendon rupture. Due to knee valgus instability (passive knee valgus showed 20°), the thickness of the tibial insert was adjusted from 11 mm to 15 mm, resulting in improved valgus instability. The scarring region of the patellar tendon was resected to 10 mm in length, and the tendon was repaired using an ultra-high molecular weight polyethylene cable (Nesplon cable) and Krackow suture. The repair was secured by making a figure-8 pattern with the cable. After the reconstruction surgery, the knee was immobilized at 0° extension for 3 weeks, followed by the initiation of range-of-motion exercises. Three months later, the extension lag was reduced to -15°, and the patient could walk without orthosis and reported neither instability nor surgical site infection at 8 months after the surgery. In conclusion, this case is notable due to the rarity of incomplete (stretched out) patellar tendon rupture post-total knee arthroplasty and demonstrates the effectiveness of Nesplon cable with Krackow suture in reconstruction surgery.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815391","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

Refractory skin ulcers and afebrile bacteremia with Staphylococcus aureus in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis: a case report.

IF 0.9

Modern rheumatology case reports Pub Date : 2024-12-12 DOI: 10.1093/mrcr/rxae082

Tokio Katakura, Tsuyoshi Shirai, Yusho Ishii, Hiroko Sato, Hiroshi Fujii

{"title":"Refractory skin ulcers and afebrile bacteremia with Staphylococcus aureus in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis: a case report.","authors":"Tokio Katakura, Tsuyoshi Shirai, Yusho Ishii, Hiroko Sato, Hiroshi Fujii","doi":"10.1093/mrcr/rxae082","DOIUrl":"https://doi.org/10.1093/mrcr/rxae082","url":null,"abstract":"The skin ulcers sometimes appear in patients with anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive dermatomyositis (DM), and usually associates with disease activity. Here, we report a case of 41-year-old woman with anti-MDA5 antibody-positive DM, who developed refractory skin ulcers during the remission induction therapy, which were proven to be associated with clinically silent Staphylococcus aureus bacteremia with septic thrombi in her lung. The patient was referred to our hospital for the treatment of amyopathic DM with interstitial lung disease. Anti-MDA5-positive DM was diagnosed, and she was treated with triple therapy combined with tofacitinib because poor prognostic factors existed. Although the remission induction therapy improved most of symptoms, she developed erythematous rashes with ulcer on her left auricle and forearm, which were refractory to topical immunosuppressive medications. Despite the absence of systemic symptoms or elevated inflammatory markers, blood and wound cultures revealed S. aureus, and a new cavitary lesion was detected in her left lung. Subsequent antibiotic therapy resolved both the cutaneous and pulmonary lesions. This case highlights the importance of considering bacteremia and performing blood cultures when DM-related skin ulcers resist conventional treatments, even without fever during immunosuppressive therapy.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815392","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

Above-Knee Amputation in a 69-Year-Old Male with Gouty Tophi Complicated by Necrotizing Fasciitis - A Case Report.

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Modern rheumatology case reports Pub Date : 2024-12-10 DOI: 10.1093/mrcr/rxae080

Metasebia Worku Abebe, Abigya Aschalew Asfaw

引用次数: 0