Modern rheumatology case reports最新文献

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Multiple lymphadenopathies in eosinophilic granulomatosis with polyangiitis: differentiating from IgG4-related lymphadenopathy. 嗜酸性粒细胞肉芽肿伴多血管炎的多发性淋巴结病:与 IgG4 相关淋巴结病相鉴别。
Modern rheumatology case reports Pub Date : 2024-04-25 DOI: 10.1093/mrcr/rxae022
Jun-ichi Kurashina, Y. Shimojima, D. Kishida, T. Ichikawa, Takeshi Uehara, Yoshiki Sekijima
{"title":"Multiple lymphadenopathies in eosinophilic granulomatosis with polyangiitis: differentiating from IgG4-related lymphadenopathy.","authors":"Jun-ichi Kurashina, Y. Shimojima, D. Kishida, T. Ichikawa, Takeshi Uehara, Yoshiki Sekijima","doi":"10.1093/mrcr/rxae022","DOIUrl":"https://doi.org/10.1093/mrcr/rxae022","url":null,"abstract":"We report a case of eosinophilic granulomatosis with polyangiitis (EGPA) in a 75-year-old man who developed mononeuritis multiplex and purpura in the lower legs concomitantly presenting with lymphadenopathies. Biopsied lymph node tissue pathologically demonstrated fibrinoid necrotizing vasculitis with perivascular eosinophil infiltration, resulting in EGPA diagnosis. Additionally, abundant IgG4-positive plasma cell infiltration exhibiting >70% IgG4/IgG ratio, without storiform pattern fibrosis and obliterative phlebitis, was observed in the biopsied lymph node. Clinical improvement was observed after corticosteroid therapy. IgG4-related lymphadenopathy (IgG4-LAD) has been defined as a distinct clinical category regardless of fulfilling IgG4-related disease classification criteria. However, some autoimmune diseases, including EGPA, can develop lymphadenopathy pathologically similar to IgG4-LAD.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2024-04-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140654773","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
Development of anti-MDA5 autoantibody-positive dermatomyositis following the use of etanercept biosimilar in rheumatoid arthritis. 类风湿性关节炎患者使用依那西普生物类似物后出现抗MDA5自身抗体阳性皮肌炎。
Modern rheumatology case reports Pub Date : 2024-04-10 DOI: 10.1093/mrcr/rxae013
Soshi Okazaki, T. Shirai, Hiroko Sato, T. Ishii, Hiroshi Fujii
{"title":"Development of anti-MDA5 autoantibody-positive dermatomyositis following the use of etanercept biosimilar in rheumatoid arthritis.","authors":"Soshi Okazaki, T. Shirai, Hiroko Sato, T. Ishii, Hiroshi Fujii","doi":"10.1093/mrcr/rxae013","DOIUrl":"https://doi.org/10.1093/mrcr/rxae013","url":null,"abstract":"The induction of autoimmune diseases during tumor necrosis factor-alpha inhibitor (TNFi) usage has been described. Herein, we report a rare case of a 49-year-old woman with anti-melanoma differentiation-associated gene 5 (MDA5) antibody (Ab)-positive dermatomyositis (DM), which developed five weeks after the introduction of an etanercept biosimilar to rheumatoid arthritis (RA). Four of the five known cases, including ours, of anti-MDA5Ab positive DM complicated with RA revealed anti-MDA5Ab positive DM following TNFi usage. When patients with RA are diagnosed with interstitial lung disease during TNFi usage, anti-MDA5 Ab-positive DM could be a differential diagnosis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2024-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140716324","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
Efficacy of anifrolumab in long-term intractable alopecia due to discoid lupus erythematosus. 安非他酮对盘状红斑狼疮引起的长期顽固性脱发的疗效。
Modern rheumatology case reports Pub Date : 2024-04-10 DOI: 10.1093/mrcr/rxae018
N. Azuma, Masaru Natsuaki, Naoaki Hashimoto, T. Abe, Suzu Ueda, Yuko Ohno, Masatoshi Jinnin, Kiyoshi Matsui
{"title":"Efficacy of anifrolumab in long-term intractable alopecia due to discoid lupus erythematosus.","authors":"N. Azuma, Masaru Natsuaki, Naoaki Hashimoto, T. Abe, Suzu Ueda, Yuko Ohno, Masatoshi Jinnin, Kiyoshi Matsui","doi":"10.1093/mrcr/rxae018","DOIUrl":"https://doi.org/10.1093/mrcr/rxae018","url":null,"abstract":"Alopecia associated with lupus erythematosus is broadly classified into reversible nonscarring alopecia seen in the acute phase, such as worsening of systemic lupus erythematosus (SLE) and cicatricial alopecia seen in chronic cutaneous lupus erythematosus represented by discoid lupus erythematosus (DLE). In DLE-induced alopecia, early therapeutic intervention before developing scarring alopecia is important, but the condition is often resistant to conventional treatment. Anifrolumab (ANI), a novel therapeutic agent for SLE that inhibits type I interferon activity, has been shown to be effective against acute skin lesions, including alopecia, in patients with SLE. However, there are very few reports on the effect of ANI on alopecia due to DLE. We report on a 27-year-old Japanese woman with SLE whose alopecia due to chronic DLE was refractory to topical therapy and systemic therapy with oral glucocorticoid, multiple immunosuppressants and belimumab for approximately 8 years after onset, and whose alopecia improved with ANI. ANI can be considered to be an effective treatment option in lupus patients presenting with alopecia due to DLE, even in the chronic refractory stage.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2024-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140719034","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 erythema nodosum with salazosulfapyridine in a 9-year-old patient with chronic non-bacterial osteomyelitis. 用柳氮磺吡啶成功治疗一名 9 岁慢性非细菌性骨髓炎患者的结节性红斑。
Modern rheumatology case reports Pub Date : 2024-04-10 DOI: 10.1093/mrcr/rxae020
Masaki Shimomura, Yuka Okura, Yutaka Takahashi, Ichiro Kobayashi
{"title":"Successful treatment of erythema nodosum with salazosulfapyridine in a 9-year-old patient with chronic non-bacterial osteomyelitis.","authors":"Masaki Shimomura, Yuka Okura, Yutaka Takahashi, Ichiro Kobayashi","doi":"10.1093/mrcr/rxae020","DOIUrl":"https://doi.org/10.1093/mrcr/rxae020","url":null,"abstract":"We report a 9-year-old Japanese girl with chronic non-bacterial osteomyelitis (CNO) accompanied by recurrent erythema nodosum (EN) which was successfully treated with salazosulfapyridine (SASP). She was referred to our hospital because of recurrent erythema on her lower extremities and persistent knee and ankle arthralgia, which had been present for approximately one year. Although naproxen, a nonsteroidal anti-inflammatory drug (NSAID), was initiated, her symptoms frequently recurred. Magnetic resonance imaging demonstrated multiple distinct high-intensity signals in the talus bones suggestive of multiple bone edemas. Additionally, a histological examination of erythematous lesions was consistent with the histopathological findings of EN. She was diagnosed as having CNO complicated by EN, and received 250 mg/day of SASP as a second-line treatment, which showed partial response of both skin and bone lesions. Following increase in the dose of SASP to 500 mg/day resulted in complete remission of her skin and bone lesions. In conclusion, our findings suggest that SASP is effective not only for CNO bone lesions but also for EN. SASP could serve as a second-line therapeutic option at least for some cases of CNO complicated by EN refractory to NSAIDs.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2024-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140719861","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
Infliximab biosimilar-induced lupus nephritis: A case report. 英夫利昔单抗生物类似物诱导的狼疮性肾炎:一例报告。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad061
Kenta Shidahara, Takayuki Katsuyama, Kei Hirose, Kazuya Matsumoto, Shoichi Nawachi, Takato Nakadoi, Yosuke Asano, Yu Katayama, Yoshia Miyawaki, Eri Katsuyama, Mariko Takano-Narazaki, Yoshinori Matsumoto, Ken-Ei Sada, Jun Wada
{"title":"Infliximab biosimilar-induced lupus nephritis: A case report.","authors":"Kenta Shidahara, Takayuki Katsuyama, Kei Hirose, Kazuya Matsumoto, Shoichi Nawachi, Takato Nakadoi, Yosuke Asano, Yu Katayama, Yoshia Miyawaki, Eri Katsuyama, Mariko Takano-Narazaki, Yoshinori Matsumoto, Ken-Ei Sada, Jun Wada","doi":"10.1093/mrcr/rxad061","DOIUrl":"10.1093/mrcr/rxad061","url":null,"abstract":"<p><p>We present a case of microhematuria, proteinuria and hypocomplementemia which developed in a 55-year-old female who was being treated with an infliximab biosimilar for rheumatoid arthritis. Renal biopsy showed lupus nephritis (ISN/RPS classification class IV + V). Treatment with the infliximab biosimilar was discontinued, and treatment with prednisolone, hydroxychloroquine and abatacept was started, resulting in clinical remission of lupus nephritis and RA. Although tumour necrosis factor-α α inhibitors are known to induce production of autoantibodies, symptoms are usually limited to skin involvement or arthritis, and renal complications are rare. Physicians should be aware of the risk of lupus nephritis and carefully monitor patients for the development of renal involvement during treatment with tumour necrosis factor-α inhibitors.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71416412","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
The first case of SARS-CoV-2-induced eosinophilic fasciitis. 首例严重急性呼吸系统综合征冠状病毒2型引起的嗜酸性筋膜炎。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad063
Hiba Boussaa, Mariem Kamoun, Saoussen Miladi, Yasmine Makhlouf, Kawther Ben Abdelghani, Alia Fazaa, Ahmed Laatar
{"title":"The first case of SARS-CoV-2-induced eosinophilic fasciitis.","authors":"Hiba Boussaa, Mariem Kamoun, Saoussen Miladi, Yasmine Makhlouf, Kawther Ben Abdelghani, Alia Fazaa, Ahmed Laatar","doi":"10.1093/mrcr/rxad063","DOIUrl":"10.1093/mrcr/rxad063","url":null,"abstract":"<p><p>Eosinophilic fasciitis (EF), also known as Shulman syndrome, is a rare auto-immune fibrosing disorder of the fascia. Etiopathogeny of EF is still unclear. Nowadays, it is widely known that severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) may induce hyper-stimulation of the immune system. Several cases with fasciitis and rhabdomyolysis induced by coronavirus disease 2019 vaccines have been reported in the literature. Herein, we report the first case of EF possibly triggered by SARS-CoV-2 infection. A 45-year-old Tunisian woman, with no medical history, presented to our department with severe widespread muscle pain noticed one month after a SARS-CoV-2 infection. Physical examination showed an induration of the skin and subcutaneous tissue of the arms, forearms and legs with a restricted joint mobility. The level of eosinophils was 430 E/mm3 (6.1%) [1-4%]. Electromyography and creatine kinase levels were normal. Myositis-related antibodies were negative. Magnetic resonance imaging of the left arm showed high-intensity signal and thickness of the fascia without evidence of muscle or bone involvement. A muscular biopsy from the right deltoid showed thickening and inflammation of the fascia. The patient received intraveinous injections of 1000 mg of methylprednisolone for 3 days with an oral relay of 1 mg/kg per day of prednisone equivalent during 4 weeks. At one-month follow-up, a significant improvement of the skin induration and myalgia was observed, with a disappearance of the biological inflammatory syndrome. This brief report suggests a potential link between SARS-CoV-2 infection and new-onset of auto-immune fasciitis.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71416414","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
Re-emphasising the importance of catheter-based angiography to differentiate polyarteritis nodosa from cutaneous arteritis: Two case reports. 再次强调导管血管造影术鉴别结节性多动脉炎和皮动脉炎的重要性:两例报告。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad064
Koji Suzuki, Mitsuhiro Akiyama, Yasushi Kondo, Shuntaro Saito, Jun Kikuchi, Hironari Hanaoka, Yuko Kaneko
{"title":"Re-emphasising the importance of catheter-based angiography to differentiate polyarteritis nodosa from cutaneous arteritis: Two case reports.","authors":"Koji Suzuki, Mitsuhiro Akiyama, Yasushi Kondo, Shuntaro Saito, Jun Kikuchi, Hironari Hanaoka, Yuko Kaneko","doi":"10.1093/mrcr/rxad064","DOIUrl":"10.1093/mrcr/rxad064","url":null,"abstract":"<p><p>Polyarteritis nodosa (PAN) is a systemic necrotising vasculitis with a poor prognosis, characterised by inflammation and necrosis of medium-sized arteries. PAN patients can present with a wide range of systemic manifestations, whereas cutaneous arteritis (CA) is a restricted manifestation to skin of the disease with a more favourable prognosis. Thus, differentiation between PAN and CA is crucial. Here, we present two cases that were initially diagnosed as CA due to the limited presence of systemic symptoms, but were finally diagnosed as PAN through catheter-based angiography. Although contrast-enhanced computed tomography and computed tomographic angiography are increasingly used to diagnose PAN, neither case had any abnormal findings on these examinations. Our cases therefore underscore that catheter-based angiography is critical for differentiation between PAN and CA, even in cases with limited systemic symptoms.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"72016589","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
Eosinophilic granulomatous with polyangiitis complicated by swelling of the oral cavity floor and cervical soft tissue as initial manifestation mimicking IgG4-related disease: A case report. 嗜酸性肉芽肿伴多血管炎,并伴有口腔底和颈部软组织肿胀,其最初表现类似IgG4相关疾病:一例报告。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad062
Tomoko Suzuki, Mayuko Moriyama, Ikuko Takano, Nobue Miyajima, Yuki Yoshioka, Manabu Honda, Masahiro Kondo, Sachiko Shokei, Asuka Araki, Kyuichi Kadota, Kunihiro Ichinose
{"title":"Eosinophilic granulomatous with polyangiitis complicated by swelling of the oral cavity floor and cervical soft tissue as initial manifestation mimicking IgG4-related disease: A case report.","authors":"Tomoko Suzuki, Mayuko Moriyama, Ikuko Takano, Nobue Miyajima, Yuki Yoshioka, Manabu Honda, Masahiro Kondo, Sachiko Shokei, Asuka Araki, Kyuichi Kadota, Kunihiro Ichinose","doi":"10.1093/mrcr/rxad062","DOIUrl":"10.1093/mrcr/rxad062","url":null,"abstract":"<p><p>Eosinophilic granulomatous polyangiitis is a systemic vasculitis associated with bronchial asthma and eosinophilic sinusitis. Here, we describe an unusual presentation of eosinophilic granulomatous polyangiitis that initially manifested as swelling of the oral cavity floor and cervical soft tissue. A 58 year-old Japanese man was diagnosed with bronchial asthma during childhood but did not receive regular medication. Prior to this presentation, he had a persistent cough for over 1 month, and a local physician diagnosed him with bronchial asthma. However, 6 months later, his cough worsened, and a blood test revealed elevated eosinophil levels. Immediately afterward, swelling of the floor of the oral cavity and cervical soft tissue developed. Cellulitis was suspected and antimicrobial treatment was initiated; however, the symptoms persisted and abdominal pain developed. An endoscopic examination revealed duodenitis and a duodenal ulcer. The patient was diagnosed with eosinophilic granulomatous polyangiitis based on three items of the 2022 American College of Rheumatology/European College of Rheumatology classification criteria: obstructive airway disease, blood eosinophil count ≥1 × 109 cells/L, and extravascular eosinophilic infiltration with a score of 10. Oral prednisolone (70 mg/day), intravenous cyclophosphamide (500 mg/m2), and subcutaneous mepolizumab (300 mg every 4 weeks) were administered. The patient's symptoms improved after these treatments, and the eosinophil count and inflammatory marker levels declined. When swelling of the oral cavity floor and cervical soft tissue following an increase in eosinophilia and allergic symptoms occurs, it is crucial to consider the likelihood of eosinophilic granulomatous polyangiitis and collaborate with otolaryngologists and dentists to ensure its prompt identification.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71416411","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
Solitary recurrence of IgG4-related giant coronary aneurysm: Case report and review of the literature focusing on treatment strategies and complications. IgG4相关巨大冠状动脉瘤的孤立性复发:病例报告和文献综述,重点关注治疗策略和并发症。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad065
Hiroyuki Kawahara, Ichiro Mizushima, Yasushi Matsumoto, Kenji Sakata, Masayuki Takamura, Dai Inoue, Satomi Kasashima, Mitsuhiro Kawano
{"title":"Solitary recurrence of IgG4-related giant coronary aneurysm: Case report and review of the literature focusing on treatment strategies and complications.","authors":"Hiroyuki Kawahara, Ichiro Mizushima, Yasushi Matsumoto, Kenji Sakata, Masayuki Takamura, Dai Inoue, Satomi Kasashima, Mitsuhiro Kawano","doi":"10.1093/mrcr/rxad065","DOIUrl":"10.1093/mrcr/rxad065","url":null,"abstract":"<p><p>Coronary periarteritis is a dangerous manifestation of IgG4-related disease, because it forms coronary artery aneurysms, which may cause sudden cardiac death. We report the case of a 78-year-old woman with IgG4-related coronary periarteritis and a coronary aneurysm, which showed progressive enlargement despite maintenance therapy for Type 1 autoimmune pancreatitis. This case was unique, in that coronary periarteritis was the only active lesion that recurred. Low-dose glucocorticoids suppressed the progression of periarterial lesions but led to rapid thinning of the aneurysmal wall and an increase in the size of mural thrombi, which pose a risk of myocardial infarction. Our systematic literature review including 98 cases of 86 articles was performed to examine its treatment strategies and complications. Among the cases in which the effect of immunosuppressive therapy could be followed radiologically, 33 of 37 (89.1%) cases showed improvement in wall thickening/periarterial soft tissue, while 6 of 13 (46.2%) showed worsening increase in the outer diameter of the coronary aneurysms. We propose a draft treatment algorithm and suggest that immunosuppressive therapy for IgG4-related coronary periarteritis with coronary aneurysms should be conducted only after the therapeutic benefit has been determined to outweigh the risks. Because coronary periarteritis can occur without other organ involvement, as in our case, all cases of IgG4-related disease require careful monitoring of coronary artery lesions.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"72016592","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
Colchicine-resistant sacroiliitis in a Japanese patient with familial Mediterranean fever. 一名患有家族性地中海热的日本患者的秋水仙碱耐药性骶管炎。
Modern rheumatology case reports Pub Date : 2023-12-29 DOI: 10.1093/mrcr/rxad050
Haruki Matsumoto, Yuya Sumichika, Kenji Saito, Shuhei Yoshida, Jumpei Temmoku, Yuya Fujita, Naoki Matsuoka, Tomoyuki Asano, Shuzo Sato, Kiyoshi Migita
{"title":"Colchicine-resistant sacroiliitis in a Japanese patient with familial Mediterranean fever.","authors":"Haruki Matsumoto, Yuya Sumichika, Kenji Saito, Shuhei Yoshida, Jumpei Temmoku, Yuya Fujita, Naoki Matsuoka, Tomoyuki Asano, Shuzo Sato, Kiyoshi Migita","doi":"10.1093/mrcr/rxad050","DOIUrl":"10.1093/mrcr/rxad050","url":null,"abstract":"<p><p>The articular involvement in patients with familial Mediterranean fever (FMF) represents a clinical characteristic of acute monoarthritis with pain and hydrarthrosis, which always resolves spontaneously. Colchicine prevents painful arthritis attacks in most FMF cases. Spondyloarthritis is rarely associated with Japanese patients with FMF. Here, we report a Japanese male patient with FMF-related axial joint involvement. A 43-year-old male Japanese patient who presented with recurrent febrile episodes with hip joint and back pain was referred to our hospital. He carried heterozygous variants in exon 2 (L110P/E148Q) of the MEFV gene. FMF was suspected, and oral administration of colchicine (1 mg/day) was initiated. Colchicine treatment improved his febrile attack with hip joint pain. He was diagnosed as having FMF based on the Tel-Hashomer diagnostic criteria for FMF since he fulfilled one major criterion (repeated febrile attack accompanied by hip joint pain) and one minor criterion (improvement with colchicine treatment). Although the human leucocyte antigen-B27 allele was not detected, sacroiliitis-related symptoms progressed despite the ongoing colchicine treatment. Salazosulphapyridine and methotrexate were administered in addition to colchicine; however, these treatments were not effective. Canakinumab treatment successfully resolved this unique aspect of sacroiliitis, and the patient was finally diagnosed with FMF-associated axial joint involvement.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71416402","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
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