Modern rheumatology case reports最新文献_第10页

Macrophage migration inhibitory factor as a potential 'missing important factor' driving inflammatory arthritis in adrenocorticotropic hormone deficiency. 巨噬细胞迁移抑制因子是促肾上腺皮质激素缺乏症引发炎症性关节炎的潜在 "缺失的重要因素"。

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Modern rheumatology case reports Pub Date : 2024-11-12 DOI: 10.1093/mrcr/rxae070

Lisa Wang, Akihiro Nakamura

引用次数: 0

A case of cryopyrin-associated periodic syndrome due to somatic mosaic mutation complicated with recurrent circinate erythematous psoriasis. 体细胞镶嵌突变引起的冷冻素相关周期综合征并发复发性环状红斑性银屑病1例。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxad067

Taiki Ando, Yoshiyuki Abe, Ken Yamaji, Ryuta Nishikomori, Naoto Tamura

{"title":"A case of cryopyrin-associated periodic syndrome due to somatic mosaic mutation complicated with recurrent circinate erythematous psoriasis.","authors":"Taiki Ando, Yoshiyuki Abe, Ken Yamaji, Ryuta Nishikomori, Naoto Tamura","doi":"10.1093/mrcr/rxad067","DOIUrl":"10.1093/mrcr/rxad067","url":null,"abstract":"Cryopyrin-associated periotic syndrome (CAPS) is a rare autoinflammatory disease caused by genetic variants in innate immunity genes. Autoinflammatory diseases, including CAPS, mediate proinflammatory cytokines such as interleukin (IL)-1 and IL-18 and result in severe systemic inflammation. A gain-of-function mutation in the NLR family pyrin domain-containing 3 (NLRP3) gene, which encodes the protein cryopyrin, was identified to be responsible for CAPS in 2001, and since then several additional pathogenic mutations have been found. Moreover, other phenotypes have been identified based on severity and symptomatology, including familial cold autoinflammatory syndrome, Muckle-Wells syndrome, and neonatal-onset multisystem inflammatory disease/chronic neurologic cutaneous articular syndrome. Prompt diagnosis of CAPS remains challenging, however, due to unspecific, extensive clinical signs, and delayed diagnosis and treatment targeting IL-1 lead to multiorgan damage. Another factor complicating diagnosis is the existence of somatic mosaic mutations in the NLRP3 gene in some cases, resulting in symptoms and clinical courses that are atypical. The frequency of somatic mosaic mutations in CAPS was estimated to be 19% in a systematic review. Psoriasis is a chronic inflammatory skin disease that affects ∼3% of the global population. Although no reports have shown complication between CAPS and psoriasis, these diseases have several similarities and potential relationships, for instance activation of T helper 17 cells in the dermis and increased NLRP3 gene expression in psoriatic skin compared with normal skin. Here, we report a case of CAPS due to a somatic mosaic mutation with recurrent circinate erythematous psoriasis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"368-372"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138465246","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

Neurofascialvascular training for the treatment of Raynaud's phenomenon: A case report. 治疗雷诺现象的神经-筋膜-血管训练：病例报告。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae026

Paolo Bertacchini

{"title":"Neurofascialvascular training for the treatment of Raynaud's phenomenon: A case report.","authors":"Paolo Bertacchini","doi":"10.1093/mrcr/rxae026","DOIUrl":"10.1093/mrcr/rxae026","url":null,"abstract":"Primary Raynaud's phenomenon (PRP) is characterised by episodic, reversible, and disabling vasospasms of the peripheral arteries. In the most severe cases, it can lead to ulceration of the fingers and toes. Neuro fascial VascularTraining (NFVT) is a novel therapeutic approach for treating PRP. NFVT aims to enhance peripheral circulation and stimulate the autonomic nervous system (ANS) by engaging multiple physiological mechanisms simultaneously. This integrated approach works to reduce vasospasms and alleviate associated symptoms through neurodynamic and myofascial interventions. A 54-year-old woman, who has experienced pain and hypoesthesia in her hands for 9 years, received a diagnosis of PRP without systemic sclerosis in 2014. The patient reported daily colour changes in her fingers, along with pain and a temporary decrease in tactile sensitivity. The patient engaged in ten 30-minute exercise sessions, and the clinical outcomes were assessed based on several parameters. These included the frequency and duration of vasospastic attacks, evaluated using the Raynaud Condition Score, as well as pain and tingling, measured through the daily Numeric Rating Scale. The Composite Autonomic Symptom Score (COMPASS 31) was utilised to assess dysautonomia, while the frequency of medication use and the Disabilities of the Arm, Shoulder, and Hand questionnaire were also considered. The results indicated a significant improvement in symptoms. NFVT improved symptoms and motor dysfunction in a patient with Raynaud's syndrome, demonstrating how NFVT can increase peripheral blood flow, stimulate the ANS, and improve symptoms in PRP.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"302-309"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140900759","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

Painful snapping of the middle finger caused by hyperplasia of the ulnar lateral band: A case report. 尺侧带增生导致的中指折断疼痛：病例报告。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxad073

Anna Matsuo, Taku Suzuki, Hiroo Kimura, Noboru Matsumura, Takuji Iwamoto, Masaya Nakamura

引用次数: 0

Improvement of active salivary gland ultrasonography findings in Sjögren's syndrome in response to short-term glucocorticoid treatment: A case report and review of the literature. 短期糖皮质激素治疗可改善斯约格伦综合征活动性唾液腺超声检查结果：病例报告和文献综述。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae002

Takashi Kida, Yutaka Kawahito, Yusuke Suzuki, Shigenori Tamaki, Ikuko Tanaka

{"title":"Improvement of active salivary gland ultrasonography findings in Sjögren's syndrome in response to short-term glucocorticoid treatment: A case report and review of the literature.","authors":"Takashi Kida, Yutaka Kawahito, Yusuke Suzuki, Shigenori Tamaki, Ikuko Tanaka","doi":"10.1093/mrcr/rxae002","DOIUrl":"10.1093/mrcr/rxae002","url":null,"abstract":"Salivary gland ultrasonography is a non-invasive imaging technique that helps in the diagnosis and assessment of disease activity in Sjögren's syndrome. However, it remains unclear whether the salivary gland ultrasonography findings are reversible in response to treatment. We present a case of a woman in her 20s who presented with parotid swelling and pain lasting for 3 months. The patient was diagnosed with anti-SS-A antibody-positive Sjögren's syndrome with active sialadenitis, and short-term glucocorticoid treatment resulted in resolution of clinical symptoms and improvement of salivary gland ultrasonography findings by the Outcome Measures in Rheumatology Clinical Trials scoring system. Notably, the anechoic/hypoechoic foci and vascular signals in the parotid and submandibular glands were reduced after treatment. Furthermore, peak systolic blood flow velocity of the facial artery entering the submandibular gland was decreased. Our case highlights that the findings of 'inflammatory' structural changes and vascularisation on salivary gland ultrasonography, including the Outcome Measures in Rheumatology Clinical Trials scoring system, reflect the disease active of Sjögren's syndrome and are reversible with treatment. Salivary gland ultrasonography has the potential to be a useful tool for monitoring treatment response and stratifying patients by disease activity in Sjögren's syndrome; therefore, further research is needed on the relationship of salivary gland ultrasonography findings with the pathophysiological mechanisms of sialadenitis and long-term clinical outcomes.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"286-290"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139473128","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

Avacopan's potential to decrease MPO-ANCA titres concurrent with ameliorated activity in ANCA-associated vasculitis. Avacopan 可降低 MPO-ANCA 滴度，同时改善 ANCA 相关性血管炎的活动。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae016

Tomoki Taniguchi, Ryosuke Hiwa, Mikihito Shoji, Eriho Yamaguchi, Mirei Shirakashi, Hideo Onizawa, Hideaki Tsuji, Koji Kitagori, Ran Nakashima, Shuji Akizuki, Akira Onishi, Hajime Yoshifuji, Masao Tanaka, Akio Morinobu

{"title":"Avacopan's potential to decrease MPO-ANCA titres concurrent with ameliorated activity in ANCA-associated vasculitis.","authors":"Tomoki Taniguchi, Ryosuke Hiwa, Mikihito Shoji, Eriho Yamaguchi, Mirei Shirakashi, Hideo Onizawa, Hideaki Tsuji, Koji Kitagori, Ran Nakashima, Shuji Akizuki, Akira Onishi, Hajime Yoshifuji, Masao Tanaka, Akio Morinobu","doi":"10.1093/mrcr/rxae016","DOIUrl":"10.1093/mrcr/rxae016","url":null,"abstract":"Avacopan, an orally administered C5a receptor antagonist, is effective in microscopic polyangiitis via the inhibition of neutrophil priming induced by C5a. However, the exact effect of avacopan on the production of myeloperoxidase antineutrophil cytoplasmic antibody (MPO-ANCA) is yet to be clearly established. This report presents a microscopic polyangiitis patient without major organ damage where high levels of MPO-ANCA persisted with high-dose steroid therapy and azathioprine, but the addition of avacopan led to a reduction in MPO-ANCA titres. The present case implies that avacopan-mediated inhibition of C5a may lead to a reduction in MPO-ANCA levels, thereby potentially ameliorating the pathophysiology of ANCA-associated vasculitis. Nevertheless, the impact of avacopan on MPO-ANCA production cannot be asserted solely based on this report; therefore, further examination is necessary through subgroup analysis using data from larger-scale studies.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"314-317"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140873989","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

Hypothermia, bradycardia, and hypotension during glucocorticoid or cyclosporine A therapy in a boy with Kikuchi-Fujimoto disease. 一名患有菊池-藤本氏病的男孩在接受糖皮质激素或环孢素 A 治疗期间出现低体温、心动过缓和低血压。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae023

Yasuyoshi Hiramatsu, Kazuki Takahashi, Masaki Shimomura, Kota Taniguchi, Yuka Okura, Mitsuru Nawate, Yutaka Takahashi, Ichiro Kobayashi

{"title":"Hypothermia, bradycardia, and hypotension during glucocorticoid or cyclosporine A therapy in a boy with Kikuchi-Fujimoto disease.","authors":"Yasuyoshi Hiramatsu, Kazuki Takahashi, Masaki Shimomura, Kota Taniguchi, Yuka Okura, Mitsuru Nawate, Yutaka Takahashi, Ichiro Kobayashi","doi":"10.1093/mrcr/rxae023","DOIUrl":"10.1093/mrcr/rxae023","url":null,"abstract":"Kikuchi-Fujimoto disease (KFD) is an inflammatory disease of unknown aetiology characterised by fever and cervical lymphadenopathy. Although KFD is a self-limiting disease, patients with severe or long-lasting course require glucocorticoid therapy. We presently report a 17-year-old boy with KFD who had seven relapses since the onset at 4 years old. He suffered from hypothermia, bradycardia, and hypotension during the treatment with prednisolone or methylprednisolone. All of his vital signs recovered after cessation of the drug in addition to fluid replacement and warming. Thus, glucocorticoid was effective but could not be continued because of the adverse event. Although hypothermia developed during the treatment with 5 mg/kg/day of cyclosporine A (CsA) at his second relapse, he was successfully treated with lower-dose CsA (3 mg/kg/day). Thereafter, he had five relapses of KFD until the age of 12 years and was treated by 1.3-2.5 mg/kg/day of CsA. Hypothermia accompanied by bradycardia and hypotension developed soon after concomitant administration of ibuprofen at his fifth and sixth relapses even during low-dose CsA therapy. Conclusively, glucocorticoid, standard dose of CsA, or concomitant use of non-steroidal anti-inflammatory drugs may cause hypothermia, bradycardia, and hypotension and needs special attention. Low-dose CsA could be a choice for such cases with KFD.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"352-356"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141082923","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

Long-term clinical course of two rare cases of synovitis-acne-pustulosis-hyperostosis-osteomyelitis syndrome involving only unilateral femur. 两例仅累及单侧股骨的滑膜炎-痤疮-脓疱病-骨质增生-骨髓炎综合征罕见病例的长期临床过程。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae024

Hiroki Ito, Yuji Hirano

{"title":"Long-term clinical course of two rare cases of synovitis-acne-pustulosis-hyperostosis-osteomyelitis syndrome involving only unilateral femur.","authors":"Hiroki Ito, Yuji Hirano","doi":"10.1093/mrcr/rxae024","DOIUrl":"10.1093/mrcr/rxae024","url":null,"abstract":"Synovitis-acne-pustulosis-hyperostosis-osteomyelitis (SAPHO) syndrome is characterised by aseptic osteitis and is often complicated by pustular dermatitis, such as palmoplantar pustulosis or acne. Although bone lesions are most found in the anterior thoracic region or spine, femoral lesions are not well documented in the literature. There is no established treatment for this condition, and few reports have described its long-term course. Here, we describe two cases of SAPHO syndrome involving the femur and discuss their long-term follow-up. A 40-year-old man (Case 1) presented with right thigh pain. Fifteen years after the initial diagnosis, the pain could be controlled with minomycin, salazosulfapyridine, and methotrexate. X-rays of the femur showed gradual cortical thickening. Although there were waves of pain, it gradually improved with the adjustment of drugs 25 years following the initial diagnosis. A 35-year-old man (Case 2) with right thigh pain was prescribed salazosulfapyridine and methotrexate; however, these were ineffective. Alendronate and guselkumab also proved ineffective. Ultimately, infliximab was started 9 years following disease onset, and pain became manageable. X-rays of the femur showed cortical thickening. SAPHO syndrome can be managed with drug therapies, such as nonsteroidal anti-inflammatory drugs, methotrexate, and conventional synthetic disease-modifying antirheumatic drugs; however, there are occasional treatment-resistant cases.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"373-377"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140924120","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

Reverse V-shaped osteotomy for ankylosing rocker-bottom foot deformity in patients with rheumatoid arthritis: A report of three cases. 针对类风湿性关节炎患者强直性摇椅底足畸形的反向 V 型截骨术。3例病例报告。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae005

Takaaki Noguchi, Makoto Hirao, Kosuke Ebina, Yuki Etani, Gensuke Okamura, Hideki Tsuboi, Atsushi Goshima, Seiji Okada, Jun Hashimoto

引用次数: 0

A case of juvenile-onset ankylosing spondylitis effectively treated with tumour necrosis factor-alpha inhibitor agents. 一例使用肿瘤坏死因子-α抑制剂有效治疗的幼年强直性脊柱炎病例。

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Modern rheumatology case reports Pub Date : 2024-07-08 DOI: 10.1093/mrcr/rxae006

Akira Sakaguchi, Naoki Kondo, Rika Kakutani, Eiji Kinoshita, Yasufumi Kijima, Hiroyuki Kawashima

{"title":"A case of juvenile-onset ankylosing spondylitis effectively treated with tumour necrosis factor-alpha inhibitor agents.","authors":"Akira Sakaguchi, Naoki Kondo, Rika Kakutani, Eiji Kinoshita, Yasufumi Kijima, Hiroyuki Kawashima","doi":"10.1093/mrcr/rxae006","DOIUrl":"10.1093/mrcr/rxae006","url":null,"abstract":"A 15-year-old girl had experienced hip pain at 11 years of age. At 15 years of age, the patient complained of persistent generalised pain. Her rheumatoid factor and serum matrix metalloproteinase-3 levels were below standard values; there were no inflammatory responses, and the human leukocyte antigen test was negative for B27 and positive for B52 and B62. The bath ankylosing spondylitis disease activity index (BASDAI) value was 8.0 at the time of induction and 3.1 at 6 months after the introduction of adalimumab (at a dose of 40 mg). The BASDAI value improved with an increase in the dose of adalimumab to 80 mg at 8 months after the initial introduction of adalimumab (at 40 mg), although it remained at 4.8 at 16 months after the dose increase. The BASDAI value was 2.6 at 6 months, 2.7 at 1 year, and 1.8 at 1.5 years after the introduction of infliximab, indicating that the patient had progressed well without any adverse events. Based on this case, juvenile ankylosing spondylitis is a differential diagnosis for low back pain and generalised pain since childhood. Tumour necrosis factor (TNF) inhibitors were promptly introduced in this case, although it took 4 years from the initial presentation. TNF inhibitors were effective in treating juvenile ankylosing spondylitis in the present case without any adverse events. This case is notable because juvenile onset ankylosing spondylitis is one of the reasons for severe lumbago since childhood and because TNF inhibitors were administered promptly after diagnosis.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":"259-263"},"PeriodicalIF":0.9,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139725470","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