Modern rheumatology case reports最新文献_第2页

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 : 2025-03-17 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":"https://doi.org/10.1093/mrcr/rxaf019","url":null,"abstract":"Sarcoidosis is an immune-mediated systemic disease characterized by the presence of non-caseating granulomas in various parts of the body in the absence of another defined etiology. Neurologic involvement (neurosarcoidosis), 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 neurosarcoidosis was made once other neurovascular, infectious, metabolic, tumor-related, and immune-mediated etiologies were ruled out. Neurosarcoidosis 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. Neurosarcoidosis 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":"2025-03-17","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}

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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 : 2025-03-14 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 : 2025-03-12 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":"https://doi.org/10.1093/mrcr/rxaf004","url":null,"abstract":"Calcinosis is an intractable condition in juvenile dermatomyositis. 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 juvenile dermatomyositis. The patient with anti-nuclear matrix protein 2 autoantibody suffered from refractory juvenile dermatomyositis 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 six 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 juvenile dermatomyositis is getting complete control of the inflammation. Our case report suggests that abatacept may contribute to the improvement of calcinosis in juvenile dermatomyositis, at least as part of combination therapy with mycophenolate mofetil.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-03-12","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}

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Necrotizing Renal Vasculitis Associated with Sarcoidosis in a Patient with Esophageal Cancer: A Case Report. 食管癌患者坏死性肾血管炎合并结节病1例报告。

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Modern rheumatology case reports Pub Date : 2025-03-06 DOI: 10.1093/mrcr/rxaf018

Tsuneo Sasai, Ryosuke Hiwa, Shion Kachi, Yoko Shimizu, Shinya Yamamoto, Yuki Teramoto, Mirei Shirakashi, Hideaki Tsuji, Shuji Akizuki, Ran Nakashima, Hajime Yoshifuji, Motoko Yanagita, Akio Morinobu

{"title":"Necrotizing Renal Vasculitis Associated with Sarcoidosis in a Patient with Esophageal Cancer: A Case Report.","authors":"Tsuneo Sasai, Ryosuke Hiwa, Shion Kachi, Yoko Shimizu, Shinya Yamamoto, Yuki Teramoto, Mirei Shirakashi, Hideaki Tsuji, Shuji Akizuki, Ran Nakashima, Hajime Yoshifuji, Motoko Yanagita, Akio Morinobu","doi":"10.1093/mrcr/rxaf018","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf018","url":null,"abstract":"Sarcoidosis is a multisystem disorder characterized by non-caseating granulomas, often involving the lungs and lymph nodes, but can affect nearly any organ. Renal involvement in sarcoidosis typically presents as hypercalcemia or interstitial granulomatous nephritis. Renal vasculitis, however, is an exceedingly rare manifestation. We present a case of a 74-year-old Japanese male who was diagnosed with esophageal cancer and underwent chemoradiotherapy. He presented with hypercalcemia and renal dysfunction, and laboratory tests revealed elevated serum creatinine and hypercalcemia. Fluorodeoxyglucose-positron emission tomography/computed tomography showed intense uptake in the gluteal and adductor muscles, with no recurrence of esophageal cancer. A muscle biopsy confirmed non-necrotizing granulomas. Despite correction of hypercalcemia, proteinuria and renal dysfunction persisted, prompting a renal biopsy. The biopsy revealed pauci-immune vasculitis, with fibrin deposition and destruction of the vascular elastic lamina, without granulomas. The patient was treated with corticosteroids, which led to significant improvement in renal function and proteinuria. This case highlights the rare coexistence of sarcoidosis and renal vasculitis. Thus, even in the presence of mild urinary abnormalities, renal biopsy should be considered in the diagnostic approach to sarcoidosis patients with renal dysfunction.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143569264","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}

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Successful glucocorticoid treatment case for IgG4-related long-standing optic neuropathy. 糖皮质激素治疗igg4相关性长期视神经病变成功一例。

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Modern rheumatology case reports Pub Date : 2025-03-06 DOI: 10.1093/mrcr/rxaf015

Soma Fukami, Shin-Ichiro Ohmura, Toshitaka Yukishima, Takuto Hamada, Yoshiro Otsuki, Noriyoshi Ogawa

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Systemic sclerosis complicated by azathioprine-induced iatrogenic immunodeficiency-associated lymphoproliferative disorder: A case report. 系统性硬化症合并硫唑嘌呤诱导的医源性免疫缺陷相关淋巴细胞增生性疾病1例报告。

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Modern rheumatology case reports Pub Date : 2025-03-06 DOI: 10.1093/mrcr/rxaf017

Ryota Okazaki, Genki Inui, Yoshihiro Funaki, Miyu Nishigami, Hiroki Kohno, Miki Takata, Tomoya Harada, Akira Yamasaki

{"title":"Systemic sclerosis complicated by azathioprine-induced iatrogenic immunodeficiency-associated lymphoproliferative disorder: A case report.","authors":"Ryota Okazaki, Genki Inui, Yoshihiro Funaki, Miyu Nishigami, Hiroki Kohno, Miki Takata, Tomoya Harada, Akira Yamasaki","doi":"10.1093/mrcr/rxaf017","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf017","url":null,"abstract":"Lymphoproliferative disorders are rare complications in patients with autoimmune diseases who are receiving immunosuppressive therapy. This case report describes a 74-year-old man with diffuse cutaneous systemic sclerosis, anti-RNA polymerase III antibodies, and interstitial pneumonia. The patient's condition initially improved with prednisolone and intravenous cyclophosphamide, followed by maintenance therapy with azathioprine, nintedanib, and macitentan for pulmonary hypertension. Thirty months after initiating AZA, the patient developed nodules and ulcers in the left lower jaw and philtrum. Skin biopsy confirmed diffuse large B-cell lymphoma. Discontinuation of azathioprine led to the resolution of the ulcers, and no other lesions were found. This case highlights the risk of iatrogenic immunodeficiency-associated lymphoproliferative disorders in patients with systemic sclerosis, particularly in those with anti-RNA polymerase III antibodies, who are known to have an increased risk of malignancy. Although methotrexate-associated lymphoproliferative disorders are well documented in patients with rheumatoid arthritis, this is the first reported case of azathioprine-associated lymphoproliferative disorder in systemic sclerosis. These findings emphasise the importance of close monitoring of malignancies, including lymphoproliferative disorders, in patients with systemic sclerosis undergoing immunosuppressive therapy.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143569240","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}

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SAPHO syndrome with bone destruction in the lumbar vertebral endplates: a case report. SAPHO综合征伴腰椎终板骨破坏1例报告。

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Modern rheumatology case reports Pub Date : 2025-03-06 DOI: 10.1093/mrcr/rxaf013

Shiho Nakano, Arata Nakajima, Masato Sonobe, Shinji Taniguchi, Manabu Yamada, Keiichiro Yamamoto, Yasuchika Aoki, Koichi Nakagawa

{"title":"SAPHO syndrome with bone destruction in the lumbar vertebral endplates: a case report.","authors":"Shiho Nakano, Arata Nakajima, Masato Sonobe, Shinji Taniguchi, Manabu Yamada, Keiichiro Yamamoto, Yasuchika Aoki, Koichi Nakagawa","doi":"10.1093/mrcr/rxaf013","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf013","url":null,"abstract":"SAPHO syndrome is a rare inflammatory osteoarticular disorder, which includes autoimmune diseases such as pustulotic arthro-osteitis, inflammatory bowel disease-associated spondyloarthritis, and psoriatic arthritis. There are few reports on the treatment of SAPHO syndrome that presents with bone destruction in the spine. We present a case in which adalimumab (ADA) was administered to treat destruction of the lumbar vertebral endplates caused by SAPHO syndrome. The patient was a woman in her 20s who was referred to our hospital with complaints of low back pain; acne on the face, anterior chest, and back; and sternoclavicular joint pain. Blood tests showed a mild increase in C-reactive protein but negative results for rheumatoid factor and anti-cyclic citrullinated peptide antibody. Radiographs and computed tomography (CT) images demonstrated destruction with surrounding bone sclerosis in the cranial endplates of the L4 and L5 vertebrae and the left sternoclavicular joint. The Ankylosing Spondylitis Disease Activity Score (ASDAS) was 2.05, and the Bath Ankylosing Spondylitis Functional Index (BASFI) was 3.00. Despite the use of the maximum dose of non-steroidal anti-inflammatory drugs, her symptoms did not improve, and ADA was administered at a dose of 40 mg every two weeks. After ADA administration, both the ASDAS and BASFI were immediately reduced and low disease activity or remission was maintained thereafter. After three years, the CT images showed no progression of bone destruction in the lumbar vertebrae and sternoclavicular joint, and the patient was completely free from low back pain and was able to run normally.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143569269","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}

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Successful control of recurrent MAS by canakinumab in a Sjogren syndrome patient with homozygous MEFV P369S variants, and review of literatures. canakinumab在MEFV P369S纯合子变异体干燥综合征患者中成功控制复发性MAS，并复习文献

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Modern rheumatology case reports Pub Date : 2025-03-06 DOI: 10.1093/mrcr/rxaf016

Nobuyuki Ono, Motoki Yoshimura, Toshiya Nishida, Yusuke Yamauchi, Goro Doi, Yoko Fuyuno, Motoshi Sonoda, Hiroaki Niiro

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Low-density lipoprotein apheresis for refractory lupus nephritis: A case demonstrating marked improvement in proteinuria, hematuria, and renal function. 低密度脂蛋白穿刺治疗难治性狼疮肾炎：一例显示蛋白尿、血尿和肾功能显著改善。

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Modern rheumatology case reports Pub Date : 2025-02-07 DOI: 10.1093/mrcr/rxaf009

Narumichi Iwamura, Yuta Matsukuma, Kanako Tsutsumi, Narumi Higashi, Seiya Shomura, Noriko Uesugi, Takafumi Hamashoji, Yui Arita, Takashi Deguchi, Toshiaki Nakano

{"title":"Low-density lipoprotein apheresis for refractory lupus nephritis: A case demonstrating marked improvement in proteinuria, hematuria, and renal function.","authors":"Narumichi Iwamura, Yuta Matsukuma, Kanako Tsutsumi, Narumi Higashi, Seiya Shomura, Noriko Uesugi, Takafumi Hamashoji, Yui Arita, Takashi Deguchi, Toshiaki Nakano","doi":"10.1093/mrcr/rxaf009","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf009","url":null,"abstract":"Systemic lupus erythematosus (SLE) predominantly involves the kidneys, causing lupus nephritis. Patients with diffuse proliferative lupus nephritis frequently experience poor outcomes despite advances in immunosuppressive therapies. Low-density lipoprotein apheresis (LDL-A) has been a potential therapeutic option for steroid-resistant nephrotic syndromes, but its efficacy in lupus nephritis remains unknown. Here, we report the case of a 26-year-old female patient with SLE and renal pathology society classification IV + Ⅴ (G) A/C lupus nephritis who developed refractory nephrotic syndrome, severe hematuria, and declining renal function. Initial induction therapy was insufficient. Consequently, LDL-A significantly improved proteinuria, hematuria, and renal function. The urinary protein-to-creatinine ratio decreased from 7.15 g/gCr to 0.610 g/gCr, and hematuria dropped from >100 to 10-19 erythrocytes per high-power field. Additionally, complement levels were improved and anti-double-stranded DNA antibody titers were reduced. Ascribing these improvements solely to LDL-A remains challenging, but the rapid proteinuria and hematuria reduction within 48 h indicates a substantial contribution of LDL-A to the clinical response. The effluent from LDL-A contained not only LDL cholesterol but also measurable amounts of IgG and IgM, which may have contributed to the reduction in lupus nephritis activity. This case represents the first report of a marked hematuria reduction following LDL-A in lupus nephritis. LDL-A is a valuable adjunctive treatment in patients with refractory nephrotic syndrome or highly active lupus nephritis unresponsive to conventional induction therapies.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-02-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143366230","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}

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Breakthrough herpes zoster following recombinant zoster vaccinations in a rheumatoid arthritis patient receiving a Janus kinase inhibitor: A case report and literature review. 在接受Janus激酶抑制剂的类风湿性关节炎患者中，重组带状疱疹疫苗接种后出现突破性带状疱疹：一例报告和文献综述。

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Modern rheumatology case reports Pub Date : 2025-02-04 DOI: 10.1093/mrcr/rxaf012

Shunya Nagata, Naoto Yokogawa

{"title":"Breakthrough herpes zoster following recombinant zoster vaccinations in a rheumatoid arthritis patient receiving a Janus kinase inhibitor: A case report and literature review.","authors":"Shunya Nagata, Naoto Yokogawa","doi":"10.1093/mrcr/rxaf012","DOIUrl":"https://doi.org/10.1093/mrcr/rxaf012","url":null,"abstract":"The recombinant zoster vaccine (RZV) is immunologically and clinically effective in immunosuppressed patients. Though rheumatoid arthritis (RA) and the Janus kinase inhibitor (JAKi) increase the risk of herpes zoster (HZ) infection, breakthrough cases in which a HZ infection followed RZV administration are rare. We report herein a 63-year-old female patient with seropositive RA who experienced a HZ infection despite receiving the RZV. She had been receiving tocilizumab, methotrexate, and low-dose prednisolone until tocilizumab was switched to upadacitinib four weeks after two RZV administrations, which resulted in 63 weeks' remission. Her current admission was for a painful rash consisting of blisters and erythema on the right nasal alar and lips corresponding to the right V2 segment of the trigeminal nerve. HZ was diagnosed and treated for seven days with intravenous acyclovir, which alleviated the symptoms. JAKi can suppress a range of immunogenic mechanisms which underlie the efficacy of the RZV. The present patient was expected to respond favorably to the RZV because JAKi had not been administered prior to the vaccinations; however, the later start of JAKi therapy caused a breakthrough HZ infection. Immunocompromised patients have a higher risk of severe HZ, including the disseminated form, but breakthrough cases are relatively rare. The RZV is recommended as a prophylaxis against HZ as well as means of mitigating its severity when it does occur.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2025-02-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143124196","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