{"title":"Glucocorticoid sparing effect on Adalimumab for refractory Pyoderma gangrenosum in patients with Rheumatoid Arthritis: A Case series.","authors":"Shin-Ichiro Ohmura, Naomi Morishita, Haruka Yonezawa, Toshitaka Yukishima, Yusuke Ohkubo","doi":"10.1093/mrcr/rxae075","DOIUrl":"https://doi.org/10.1093/mrcr/rxae075","url":null,"abstract":"<p><p>Pyoderma gangrenosum (PG) is a rare chronic skin disease characterized by painful skin ulcers. There are no treatment guidelines for PG; however, systemic treatment is often administered. Recently, adalimumab (ADA), a fully human monoclonal antibody against tumor necrosis factor, was approved for the treatment of refractory PG in Japan. However, data are limited, and it is not clear whether ADA has the same effect on the treatment of PG in patients with systemic rheumatic disease (SRD), including rheumatoid arthritis (RA). In addition, the glucocorticoid-sparing effect of ADA in SRD patients with PG has not yet been clarified. Herein, we present two successful cases of RA with glucocorticoid-refractory PG on ADA treatment. Our report suggests that ADA may have a glucocorticoid-sparing effect on refractory PG in patients with RA.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142809000","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}
{"title":"Seronegative rheumatoid arthritis found in an 85-year-old woman during preoperative investigations for knee replacement surgery for osteoarthritis.","authors":"Haruka Nakano, Makoto Hirao, Dong Seop Kim, Gensuke Okamura, Yoshihiko Hoshida, Takaaki Noguchi, Yuki Etani, Kosuke Ebina, Seiji Okada, Jun Hashimoto, Shiro Ohshima","doi":"10.1093/mrcr/rxae076","DOIUrl":"https://doi.org/10.1093/mrcr/rxae076","url":null,"abstract":"<p><p>This report describes the case of an 85-year-old woman who was found to have seronegative rheumatoid arthritis during preoperative investigations for knee replacement surgery for osteoarthritis. Follow-up for knee osteoarthritis was continued for many years without any symptoms involving other joints. Before total knee arthroplasty for osteoarthritis, preoperative investigations revealed a highly inflammatory state. After differential diagnosis, seronegative rheumatoid arthritis was diagnosed. Even in preoperative investigations before total knee arthroplasty for very old patients with osteoarthritis, the possibility of rheumatoid arthritis developing should always be kept in mind.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808388","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}
{"title":"Muscle vasculitis in patients with polymyalgia rheumatica; three case series.","authors":"Haruka Moriya, Yuichiro Fujieda, Yuta Inoue, Kenichi Miyamoto, Mamiko Anada, Daiki Tanaka, Akihiko Kudo, Megumi Abe, Azusa Nagai, Ryo Hisada, Michihito Kono, Masaru Kato, Olga Amengual, Yoshihiro Matsuno, Ichiro Yabe, Tatsuya Atsumi","doi":"10.1093/mrcr/rxae072","DOIUrl":"https://doi.org/10.1093/mrcr/rxae072","url":null,"abstract":"<p><p>Polymyalgia rheumatica (PMR) is a common inflammatory disorder characterized by myalgia/stiffness in proximal hip and shoulder girdle, elevated C reactive protein (CRP) and erythrocyte sedimentation rate (ESR), but its pathogenesis is not fully elucidated. We report three cases of PMR who do not respond adequately to standard treatment. Those patients had typical symptoms of myalgia and muscle weakness, with elevated CRP in absence of creatine kinase elevation. Muscle specimen showed the findings of vasculitis in all cases, therefore muscular limited vasculitis (MLV) may be an underlying pathology in PMR in those refractory cases.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808186","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}
Takeshi Mochizuki, Naoko Otani, Mari Ando, Ryo Hiroshima, Koichiro Yano, Katsunori Ikari, Ken Okazaki
{"title":"Imaging using diffusion-weighted whole-body imaging with background signal suppression for methotrexate-associated lymphoproliferative disorder: A case report.","authors":"Takeshi Mochizuki, Naoko Otani, Mari Ando, Ryo Hiroshima, Koichiro Yano, Katsunori Ikari, Ken Okazaki","doi":"10.1093/mrcr/rxae078","DOIUrl":"https://doi.org/10.1093/mrcr/rxae078","url":null,"abstract":"<p><p>Patients with rheumatoid arthritis (RA) receiving immunosuppressive therapy including methotrexate (MTX), are at risk of developing lymphoproliferative disorder (LPD). Herein, we report the case of a 61-year-old man who has been treated with MTX and sulfasalazine for seropositive RA since the age of 52. He underwent diffusion-weighted whole-body imaging with background signal suppression (DWIBS), which revealed high-intensity lesions in the affected lymph nodes of the cervical, clavicular, and axillary regions. Follow-up DWIBS after MTX withdrawal showed the suppression or disappearance of the high-intensity lesions. This case demonstrates the potential of DWIBS as a new standard imaging modality for MTX-LPD in patients with RA in clinical practice.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808199","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}
{"title":"Paradoxical pyoderma gangrenosum secondary to adalimumab successfully treated with methotrexate: a case report.","authors":"Kristy El Morr, Mohamad Ali Rida","doi":"10.1093/mrcr/rxae073","DOIUrl":"https://doi.org/10.1093/mrcr/rxae073","url":null,"abstract":"<p><p>Pyoderma gangrenosum (PG) is a rare inflammatory skin disorder, categorized under neutrophilic dermatoses. It can be idiopathic or associated with underlying conditions like inflammatory bowel disease, autoimmune diseases, and certain cancers. Some medications, including TNF-alpha inhibitors like adalimumab, can also induce this paradoxical reaction. We describe the case of a 19 year old male with adalimumab induced PG which was successfully treated with methotrexate, contributing to the understanding of drug-induced PG and alternative treatment strategies.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808382","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}
{"title":"An autopsy of a patient with polyarteritis nodosa who developed fatal intestinal perforation accompanied by systemic multiple aneurysms and arterial thrombosis.","authors":"Nao Tsugita, Ippei Miyagawa, Masanobu Ueno, Miyabi Takahashi, Shumpei Kosaka, Shingo Nakayamada, Yoshiya Tanaka","doi":"10.1093/mrcr/rxae079","DOIUrl":"https://doi.org/10.1093/mrcr/rxae079","url":null,"abstract":"<p><p>The patient was a 48-year-old man who had developed acute myocardial infarction 3 years earlier. He started experiencing recurrent attacks of abdominal pain 2 years earlier. One month before the presentation, he developed perforative peritonitis, which was treated with right hemicolectomy. Preoperative computed tomography revealed systemic thrombotic aneurysms and fibrinoid necrotizing vasculitis was detected in the vessels in the serosa of the resected intestinal specimen. These findings led to a diagnosis of polyarteritis nodosa. Despite the start of remission induction therapy with high-dose glucocorticoid and intermittent intravenous cyclophosphamide, the effect of immunosuppressive therapy was limited. Approximately 1 month after treatment initiation, he died from small intestinal perforation. Polyarteritis nodosa often exhibits nonspecific clinical symptoms, which make an early diagnosis difficult in some cases. Although the prognosis depends on the presence of ischemic lesions due to a ruptured aneurysm or intra-aneurysmal thrombi, it is not rare for the diagnosis to be made following acute myocardial infarction or acute abdominal pain. In young patients with ischemic organ dysfunction without any arteriosclerotic lesions at low risk of developing cardiovascular events, early diagnosis can be made by performing a whole-body examination with a differential diagnosis of polyarteritis nodosa.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808998","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}
{"title":"Pericardial artery lymphoma in a patient with dermatomyositis: A case report.","authors":"Kei Hirose, Takayuki Katsuyama, Natsuki Kubota, Yuya Terajima, Kazuya Matsumoto, Kenta Shidahara, Takato Nakadoi, Shoichi Nawachi, Yu Katayama, Yoshia Miyawaki, Eri Katsuyama, Mariko Narazaki-Takano, Yoshinori Matsumoto, Noboru Asada, Ken-Ei Sada, Jun Wada","doi":"10.1093/mrcr/rxae065","DOIUrl":"https://doi.org/10.1093/mrcr/rxae065","url":null,"abstract":"<p><p>A woman in her 60s presented with erythema on both elbows, dyspnea on exertion, discomfort, and swelling of the left upper extremity, followed by swelling and myalgia of the right upper and lower extremities. She was diagnosed with anti-TIF1γ antibody-positive dermatomyositis with interstitial pneumonia. The initial screening tests for malignant diseases including contrast-enhanced CT, upper and lower endoscopy, and gynecological examination did not reveal any obvious abnormalities. The patient experienced two recurrent episodes of muscle weakness and dysphagia during treatment with intravenous glucocorticoids and cyclophosphamide. Five months after diagnosis, a bone marrow biopsy and positron emission tomography- computed tomography (PET-CT) scan revealed a coronary malignant lymphoma with suspected systemic metastasis. Although chemotherapy was initiated, the patient ultimately succumbed to alveolar hemorrhage. Coronary lymphoma is very rare and there has been no report of a cases associated with myositis. PET-CT may be useful for searching malignancy in anti-TIF1γ antibody-positive dermatomyositis cases of recurrent relapse.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-11-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142712340","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}
{"title":"Persistent Liver Injury Following Avacopan Discontinuation in Antineutrophil Cytoplasmic Antibody-Associated Vasculitis: A Rare Case of Liver Biopsy in the Chronic Phase of Liver Injury.","authors":"Hiroyuki Yamaguchi, Nayu Fujii, Hideki Shimizu","doi":"10.1093/mrcr/rxae071","DOIUrl":"https://doi.org/10.1093/mrcr/rxae071","url":null,"abstract":"<p><p>Drug-induced liver injury (DILI) is a major concern associated with the use of avacopan, a new therapeutic agent for antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV). Here, we report a rare instance of liver biopsy performed for persistent liver injury following discontinuation of avacopan in an elderly woman with microscopic polyangiitis (MPA). The patient developed cholestatic liver injury after 2 months of avacopan therapy, which led to drug cessation. Despite the discontinuation of avacopan followed by supportive care, the liver enzyme levels remained elevated, necessitating liver biopsy. Histological examination revealed residual inflammation and focal necrosis around the portal vein, indicating ongoing liver injury despite avacopan withdrawal. This case report highlights the hepatotoxic potential of avacopan, the importance of vigilant liver function monitoring, and the value of liver biopsy in the chronic phase of avacopan-induced liver injury. Further research is required to elucidate avacopan hepatotoxicity mechanisms and identify potential risk factors for DILI in patients with AAV.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-11-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142645345","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}
{"title":"Macrophage migration inhibitory factor as a potential 'missing important factor' driving inflammatory arthritis in adrenocorticotropic hormone deficiency.","authors":"Lisa Wang, Akihiro Nakamura","doi":"10.1093/mrcr/rxae070","DOIUrl":"https://doi.org/10.1093/mrcr/rxae070","url":null,"abstract":"","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-11-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142831592","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}
Sepehr Khosravi, Babak Zamani, Mohammad Reza Motamed, Fahimeh H Akhoundi
{"title":"Adalimumab induced CNS demyelination in a patient with multiple pre-existing autoimmune diseases; treatment contemplation.","authors":"Sepehr Khosravi, Babak Zamani, Mohammad Reza Motamed, Fahimeh H Akhoundi","doi":"10.1093/mrcr/rxae069","DOIUrl":"10.1093/mrcr/rxae069","url":null,"abstract":"<p><p>Anti-TNF agents like Adalimumab are safe and effective for rheumatologic disorders, but they have been reported to cause demyelinating diseases like Multiple Sclerosis (MS). A 47-year-old woman with left hand weakness was evaluated. She had a previous medical history of Intermediate Uveitis (IU) and Rheumatoid arthritis (RA). anti-TNF-α treatment was initiated with adalimumab two years before this visit. MRI showed numerous T2-hyperintense lesions in bilateral periventricular, juxtacortical, and subcortical areas, which were absent in her scan before anti-TNF therapy. She was diagnosed with MS, and Adalimumab was discontinued. IV glucocorticoids (GCs) were administered with marked improvement. She was then started on anti-CD20 therapy with Rituximab. A year later, she was symptom-free, and her follow-up Brain MRI showed no new lesions. The link between MS and TNF-α inhibitors is poorly understood, but several hypotheses have been proposed. Discontinuing anti-TNF therapy alone may not be enough to prevent further demyelinating disease activity, and it is essential to consider the necessity of starting a disease-modifying treatment. Autoimmunity plays a significant role in rheumatologic and neurological diseases, and as personalized medicine advances, understanding genetic risk is crucial for selecting appropriate therapeutic targets. A thorough evaluation of a patient's family background is recommended before therapeutic decision-making, especially in patients with multiple autoimmune disorders, and the question of whether TNF-α is a suitable therapeutic target in patients with multiple autoimmune disorders is raised.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-11-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142592336","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}