Modern rheumatology case reports最新文献_第9页

Rapid radiographic progression of axial spondyloarthritis: A case of diffuse bone marrow oedema evolving to bamboo spine over 9 years. 轴型脊椎关节炎的快速影像学进展：9年多时间内弥漫性骨髓水肿发展为竹棘1例。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag027

Hisayoshi Kai, Keigo Shirasaki, Kenya Ishizu, Koji Ohta, Tetsuya Watanabe, Keiichiro Iida

{"title":"Rapid radiographic progression of axial spondyloarthritis: A case of diffuse bone marrow oedema evolving to bamboo spine over 9 years.","authors":"Hisayoshi Kai, Keigo Shirasaki, Kenya Ishizu, Koji Ohta, Tetsuya Watanabe, Keiichiro Iida","doi":"10.1093/mrcr/rxag027","DOIUrl":"10.1093/mrcr/rxag027","url":null,"abstract":"Axial spondyloarthritis (axSpA) is a chronic inflammatory disease that primarily affects the sacroiliac joints and spine, leading to progressive structural changes. The clinical course of axSpA is heterogeneous, and cases of rapid structural progression over a relatively short period have been reported. With the advent of biologic agents, it is now possible to suppress structural damage, highlighting the importance of early diagnosis and treatment. However, reports describing the early features of rapidly progressive axSpA are limited. We report the case of a 55-year-old man who presented with low back pain following a fall. Imaging revealed a vertebral fracture at T12/L1 and a bamboo spine, and he underwent percutaneous posterior fixation. Sacroiliac joint ankylosis was also observed, and a diagnosis of ankylosing spondylitis was made. The patient had visited our hospital 9 years earlier for low back pain. At that time, magnetic resonance imaging showed diffuse bone marrow oedema across multiple vertebrae and depression of the vertebral endplates. However, the oedema was not localised to the vertebral corners, and the findings were interpreted as Schmorl's nodes with surrounding oedema. Based on the current findings, the previous imaging was retrospectively interpreted as an early sign of axSpA. This case highlights that diffuse vertebral bone marrow oedema without typical corner lesions may signal rapidly progressive axSpA. Recognition of such atypical early imaging features is critical to ensure timely diagnosis and management.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147517905","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

Erdheim-Chester disease mimicking IgG4-related disease. 模拟igg4相关疾病的厄德海姆-切斯特病。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag001

Katherine E Moore, Gabriella Venter, John P Slavotinek, Mihir D Wechalekar

{"title":"Erdheim-Chester disease mimicking IgG4-related disease.","authors":"Katherine E Moore, Gabriella Venter, John P Slavotinek, Mihir D Wechalekar","doi":"10.1093/mrcr/rxag001","DOIUrl":"10.1093/mrcr/rxag001","url":null,"abstract":"Aortitis is a rare but critical manifestation of systemic inflammatory disease, most commonly associated with large-vessel vasculitides such as Takayasu arteritis and giant cell arteritis. When accompanied by multiorgan inflammation and fibrosis, it presents a broader diagnostic challenge that includes immune-mediated conditions such as immunoglobulin G4-related disease (IgG4-RD) and rare histiocytic neoplasms. While IgG4-RD is increasingly recognised due to improved diagnostic criteria and access to immunostaining, histiocytic neoplasms such as Erdheim-Chester disease (ECD) remain underdiagnosed. ECD is a clonal histiocytic neoplasm driven by mutations in the mitogen-activated protein kinase pathway, with multisystem tissue infiltration and inflammation. Unlike IgG4-RD, ECD is amenable to targeted therapies. We present a diagnostically challenging case of a middle-aged woman referred with presumed IgG4-RD following five months of progressive retroperitoneal fibrosis, bilateral ureteric obstruction, weight loss, and fatigue. Further investigation revealed aortitis involving major branches, interstitial lung disease, and adrenal and pituitary involvement, but notably without skeletal disease. Omental biopsy confirmed ECD, with a pathogenic MAP2K1 mutation detected. This case highlights the diagnostic complexity of bone-sparing ECD, a phenotype present in fewer than five percent of cases. Its clinical mimicry of vasculitis and other inflammatory disorders can obscure the underlying neoplastic process, delaying diagnosis and access to effective treatment. In patients with systemic inflammation and inconclusive serology, early consideration of ECD is essential to guide appropriate investigations and timely therapy.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146055730","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

Microscopic polyangiitis-associated interstitial lung disease with acute exacerbation after SARS-CoV-2 infection: a case report and literature review. SARS-CoV-2感染后急性加重的显微镜下多血管炎相关间质性肺疾病1例报告及文献复习

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag035

Natsuko Esa, Shogo Matsuda, Hiroko Kuwabara, Katsumasa Oe, Ayana Okazaki, Takayasu Suzuka, Yumiko Wada, Takeshi Shoda, Takuya Kotani, Tohru Takeuchi

{"title":"Microscopic polyangiitis-associated interstitial lung disease with acute exacerbation after SARS-CoV-2 infection: a case report and literature review.","authors":"Natsuko Esa, Shogo Matsuda, Hiroko Kuwabara, Katsumasa Oe, Ayana Okazaki, Takayasu Suzuka, Yumiko Wada, Takeshi Shoda, Takuya Kotani, Tohru Takeuchi","doi":"10.1093/mrcr/rxag035","DOIUrl":"10.1093/mrcr/rxag035","url":null,"abstract":"Microscopic polyangiitis (MPA) is a type of systemic inflammatory small vessel vasculitis that is frequently accompanied by interstitial lung disease (ILD). Acute exacerbations (AE) of ILD are fatal complications in patients with MPA and can be triggered by various factors, including infections. We report a case of AE of MPA-ILD following severe acute respiratory syndrome Coronavirus 2 (SARS-CoV-2) infection. An 81-year-old male with MPA during remission maintenance therapy was hospitalised for Coronavirus disease 2019 pneumonia. Treatment for SARS-CoV-2 infection improved his respiratory symptoms and radiological findings, but his respiratory condition suddenly deteriorated with a high fever on hospital Day 52. Chest high-resolution computed tomography showed diffuse bilateral ground glass opacity, and we diagnosed AE of MPA-ILD. He was refractory to steroid pulse therapy and died on the second day after the onset of AE. Pathologic autopsy revealed hyaline membrane formation over the entire bilateral lobes, consistent with the exudative phase of the diffuse alveolar damage pattern. Our case suggests that persistent immune activation during the post-acute phase of SARS-CoV-2 infection may contribute to delayed AE of MPA-ILD with a diffuse alveolar damage pattern through macrophage-driven lung injury, given that ILD exacerbation occurred approximately 2 months after the onset of Coronavirus disease 2019 pneumonia.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147950484","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

Hip chondrolysis due to enthesitis-related juvenile idiopathic arthritis treated successfully with adalimumab: A case report. 阿达木单抗成功治疗膝炎相关幼年特发性关节炎所致髋关节软骨松解。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxaf083

Gosuke Akiyama, Hyonmin Choe, Naomi Kobayashi, Ken Kumagai, Hiroyuki Ike, Yutaka Inaba

{"title":"Hip chondrolysis due to enthesitis-related juvenile idiopathic arthritis treated successfully with adalimumab: A case report.","authors":"Gosuke Akiyama, Hyonmin Choe, Naomi Kobayashi, Ken Kumagai, Hiroyuki Ike, Yutaka Inaba","doi":"10.1093/mrcr/rxaf083","DOIUrl":"10.1093/mrcr/rxaf083","url":null,"abstract":"Coxitis with rapid hip chondrolysis in juvenile patients requires careful diagnosis and treatment. This report describes a case that was initially diagnosed as idiopathic hip chondrolysis but finally diagnosed as enthesis-related juvenile idiopathic arthritis (JIA). Case: A 15-year-old boy complained of bilateral hip pain and difficulty in walking. Initially, a diagnosis of idiopathic hip chondrolysis was made based on the imaging findings of centralised joint space narrowing on plain radiography and high-signal areas in the femoral head and acetabulum on T2-weighted fat-suppressed magnetic resonance imaging (MRI) without joint effusion. After the patient was admitted to our hospital, a diagnosis of enthesis-related JIA was made. Enthesis-related JIA was suspected based on arthritic changes in the sacroiliac joints that were detected incidentally during computed tomography and MRI. After initiating adalimumab administration, MRI revealed the disappearance of abnormalities in the acetabulum and femoral head. Moreover, the hip pain and contracture gradually improved, and the patient could return to daily activities without pain. Our report highlights the importance of awareness regarding the possibility of enthesis-related JIA in juvenile patients presenting with coxitis and rapid chondrolysis of the hip joint without joint effusion.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145795729","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

Delayed diagnosis of psoriatic arthritis presenting with infection-like finger inflammation: A case report. 以感染样手指炎症为表现的银屑病关节炎的延迟诊断1例。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag005

Sho Takahashi, Yasuhiro Kiyota, Takuji Iwamoto, Akiko Torii, Satoshi Oki, Taku Suzuki, Noboru Matsumura, Masaya Nakamura

{"title":"Delayed diagnosis of psoriatic arthritis presenting with infection-like finger inflammation: A case report.","authors":"Sho Takahashi, Yasuhiro Kiyota, Takuji Iwamoto, Akiko Torii, Satoshi Oki, Taku Suzuki, Noboru Matsumura, Masaya Nakamura","doi":"10.1093/mrcr/rxag005","DOIUrl":"10.1093/mrcr/rxag005","url":null,"abstract":"Psoriatic arthritis is a chronic inflammatory disease associated with psoriasis, and its diagnosis can be challenging owing to nonspecific symptoms, absence of reliable biomarkers, and occasional delay in skin manifestations. Herein, we report a case of psoriatic arthritis that initially presented as an acute finger inflammation mimicking infection. A 46-year-old woman developed sudden swelling and pain in the left ring finger during chemotherapy for cervical cancer. Based on the results of the physical examination, laboratory tests, and magnetic resonance imaging, pyogenic flexor tenosynovitis was suspected, and synovectomy was performed; however, bacterial and mycobacterial cultures yielded negative results. Despite the administration of antibiotics, the inflammation persisted, and she was referred to the Rheumatology Department, where she was diagnosed with reactive arthritis secondary to Chlamydia infection. Although the inflammation improved after antimicrobial therapy, the finger swelling persisted. Follow-up magnetic resonance imaging and serological testing were performed, and the patient was diagnosed with seronegative rheumatoid arthritis. Four years after onset, erythematous skin lesions appeared, and dermatological evaluation confirmed plaque psoriasis; thus, a definitive diagnosis of psoriatic arthritis was established. Disease-modifying antirheumatic drug adjustments improved symptoms, but residual 'pencil-in-cup' deformity and limited finger motion remained. This case highlights the difficulty in diagnosing psoriatic arthritis when arthritis precedes skin lesions. Clinicians should consider psoriatic arthritis in persistent or refractory arthritis and carefully monitor skin and nail changes to achieve an earlier diagnosis and prevent irreversible joint damage.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146109238","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

Septic pulmonary embolism in systemic sclerosis-associated intestinal pseudo-obstruction: A case report. 系统性硬化症相关假性肠梗阻并发脓毒性肺栓塞1例。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag017

Eri Sugawara, Haruka Iino, Keita Ninagawa

引用次数: 0

A refractory polyarthritis revealing a multicentric reticulohistiocytosis. 难治性多发性关节炎表现为多中心网状组织细胞增多症。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag021

Océane Deville-Cavellin, Angélique Nallet, Stéphane Ralandison, Aude Trinquet, Yves-Marie Pers

引用次数: 0

A case of anti-nuclear matrix protein 2 antibody-positive dermatomyositis sine dermatitis with a challenging diagnosis due to the absence of typical skin manifestations. 抗核基质蛋白2抗体阳性的原发性皮肌炎病例，由于缺乏典型的皮肤表现，诊断具有挑战性。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag023

Tatsuya Kai, Naoya Nishimura, Chika Nabeshima, Tomohiro Tsuji, Motoki Yoshimura, Sho Fujimoto, Ayako Kuwahara, Masahiro Ayano, Yasutaka Kimoto, Hidenori Ogata, Takeshi Iwasaki, Noriko Isobe, Yoshinao Oda, Hiroaki Niiro

{"title":"A case of anti-nuclear matrix protein 2 antibody-positive dermatomyositis sine dermatitis with a challenging diagnosis due to the absence of typical skin manifestations.","authors":"Tatsuya Kai, Naoya Nishimura, Chika Nabeshima, Tomohiro Tsuji, Motoki Yoshimura, Sho Fujimoto, Ayako Kuwahara, Masahiro Ayano, Yasutaka Kimoto, Hidenori Ogata, Takeshi Iwasaki, Noriko Isobe, Yoshinao Oda, Hiroaki Niiro","doi":"10.1093/mrcr/rxag023","DOIUrl":"10.1093/mrcr/rxag023","url":null,"abstract":"Dermatomyositis (DM) is classically associated with distinctive skin manifestations, such as Gottron's sign or heliotrope rash. Cases of DM without these skin manifestations - termed DM sine dermatitis (DMSD) - have recently been reported, with an increasing association with anti-nuclear matrix protein 2 (NXP-2) antibody. We report a 26-year-old Japanese man who presented with muscle weakness and myalgia but without the characteristic skin manifestations of DM. Laboratory findings showed only mildly elevated levels of myogenic enzymes, and initial screening for myositis-specific antibodies (MSAs) was negative. However, the muscle biopsy demonstrated necrotic and regenerative muscle fibres with perifascicular expression of myxovirus resistance A. The findings were consistent with DM. Further MSA testing revealed positivity for the anti-NXP-2 antibody, confirming the diagnosis of DMSD. This case presented a diagnostic challenge because of the absence of typical skin rashes and only mild elevation of myogenic enzymes. Clinicians should be aware of DMSD as a potential diagnosis, particularly in patients with muscle symptoms but lacking the characteristic DM skin manifestations. In a myositis diagnosis, proactive muscle biopsy and MSA testing, including the anti-NXP-2 antibody, are crucial for ensuring early diagnosis and treatment.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147380197","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

G-CSF-induced aortitis can resolve spontaneously: comment on the article by Shimanuki et al. g - csf诱导的主动脉炎可以自行消退：评论Shimanuki等人的文章。

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag032

Takao Nagashima

引用次数: 0

Pancytopenia and splenomegaly as uncommon manifestations in a patient with immunoglobulin G4-related disease. 免疫球蛋白g4相关疾病患者少见的全血细胞减少和脾肿大表现

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Modern rheumatology case reports Pub Date : 2026-01-06 DOI: 10.1093/mrcr/rxag008

Kojiro Ikeda, Shunichiro Hanai, Soichiro Kubota, Nakako Tanaka-Mabuchi, Mayu Yazaki, Yoshiaki Kobayashi, Daiki Nakagomi

{"title":"Pancytopenia and splenomegaly as uncommon manifestations in a patient with immunoglobulin G4-related disease.","authors":"Kojiro Ikeda, Shunichiro Hanai, Soichiro Kubota, Nakako Tanaka-Mabuchi, Mayu Yazaki, Yoshiaki Kobayashi, Daiki Nakagomi","doi":"10.1093/mrcr/rxag008","DOIUrl":"10.1093/mrcr/rxag008","url":null,"abstract":"Haematological manifestations of immunoglobulin (Ig)G4-related disease (IgG4-RD) are atypical, and few studies have addressed cytopenia in this condition. We present here a case of IgG4-RD with marked pancytopenia and splenomegaly in the absence of bone marrow abnormalities. A 67-year-old woman developed diplopia, dyspnoea, pancytopenia, and hypocomplementemia. Contrast-enhanced computed tomography showed bilateral lacrimal and submandibular gland swelling, enlargement of the cervical, mediastinal, inguinal, and para-aortic lymph nodes, splenomegaly, and granular shadows in both lungs. She had been referred to our hospital on suspicion of malignant lymphoma, but a lymph node biopsy indicated the possibility of IgG4-RD. A lacrimal gland biopsy showed infiltration of numerous lymphocytes and IgG4-positive plasma cells along with mild fibrosis. A bone marrow biopsy showed normocellular marrow with no increase in plasma cells. The IgG4-positive plasma cell count was 4 per high-power field, with an IgG4/IgG ratio of 10%. After excluding potential mimics, the diagnosis was IgG4-RD presenting with pancytopenia and splenomegaly. Prednisolone was initiated at 40 mg/day, leading to the rapid normalisation of pancytopenia, recovery of complement levels, and resolution of other organ involvement such as splenomegaly. Physicians should keep in mind that IgG4-RD can present with pancytopenia and splenomegaly.","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146088705","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