{"title":"A case of polyarteritis nodosa with severe lower limb ulcer that was treated with prednisolone and tocilizumab.","authors":"Naoto Okubo, Yuki Oba, Daisuke Ikuma, Hiroki Mizuno, Masayuki Yamanouchi, Tatsuya Suwabe, Yoshifumi Ubara, Naoki Sawa","doi":"10.1093/mrcr/rxae085","DOIUrl":"https://doi.org/10.1093/mrcr/rxae085","url":null,"abstract":"<p><p>Polyarteritis nodosa (PAN) is a rare systemic necrotizing vasculitis that can lead to the formation of refractory lower limb ulcers requiring amputation. The standard treatment for severe PAN involves combination therapy with steroids and cyclophosphamide; however, some cases prove to be challenging. Recently, case reports have described the use of biological agents for PAN treatment. We present the case of a 61-year-old Japanese man with cutaneous PAN and refractory recurrent lower-limb ulcers. In 2017, the patient was admitted to hospital because of exacerbation of a right lower limb ulcer. Despite combination therapy with corticosteroids, cyclophosphamide, and endovascular therapy, the gangrene in the right lower leg progressed, and amputation was performed. The patient was temporarily stabilized with PSL monotherapy. In 2019, new ulcers were observed on the left lower limb. Owing to steroid resistance, subcutaneous tocilizumab (162 mg/week) was initiated. Over a few months, the ulcer healed completely, and left lower limb amputation was avoided. Therefore, tocilizumab could potentially be one of the treatment options for severe cases in the future.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142824935","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":"IgG4-related disease with epithelioid granulomas: a case and a review of the literature.","authors":"Shion Kachi, Hideki Oka, Shuji Sumitomo, Shigeo Hara, Koichiro Ohmura","doi":"10.1093/mrcr/rxae083","DOIUrl":"https://doi.org/10.1093/mrcr/rxae083","url":null,"abstract":"<p><p>IgG4-related disease (IgG4-RD) is a systemic, immune-mediated, fibroinflammatory disorder that affects multiple organs. Histopathologically, the supportive findings of IgG4-RD include dense lymphocytic infiltrates, obliterative phlebitis, storiform fibrosis, and elevated numbers of IgG4-positive plasma cells. However, the presence of granulomatous inflammation is generally considered highly atypical, suggesting alternative diagnoses such as sarcoidosis and lymphoma. Here, we present a case of IgG4-RD involving granulomatous lymphadenopathy. Labial salivary gland biopsy findings were consistent with IgG4-related sialadenitis. Elevated serum IgG4 levels, hypocomplementemia, and abnormal imaging findings in the kidneys and pancreas indicated an association with IgG4-RD. The patient was treated with prednisolone, which resulted in a significant improvement in the serum IgG4 and complement levels and a notable reduction in lymph node swelling. Although granulomatous inflammation is rare, integrating clinical, serological, radiological, and pathological parameters can ensure an accurate assessment within the appropriate clinicopathological context.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142819734","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":"A case report of a 14-year-old male patient with large vessel vasculitis following COVID-19.","authors":"Hiroki Nemoto, Yoshihiro Nozaki, Takashi Matsumoto, Kaori Kiyoki, Takumi Ishiodori, Atsushi Morita, Kazuo Imagawa, Takashi Murakami, Miho Takahashi, Hironori Imai, Hidetoshi Takada","doi":"10.1093/mrcr/rxae081","DOIUrl":"https://doi.org/10.1093/mrcr/rxae081","url":null,"abstract":"<p><p>Most reported cases of large vessel vasculitis (LVV) following coronavirus disease 2019 (COVID-19) have involved adults, with paediatric cases being rare. We present the case of a 14-year-old boy who developed LVV following COVID-19. Initially, he presented with fever and cough, and nasopharyngeal polymerase chain reaction testing confirmed COVID-19. His symptoms spontaneously resolved without specific COVID-19 treatments. However, 10 days after contracting COVID-19, his fever recurred, and his inflammatory markers were significantly elevated. His condition did not meet the criteria for Kawasaki disease or multisystem inflammatory syndrome in children associated with COVID-19. Contrast-enhanced computed tomography revealed arterial wall thickening in the aorta and carotid arteries, indicative of LVV. Upon initiation of high-dose immunoglobulin therapy and aspirin, his fever subsided, and his inflammatory markers and imaging findings normalised. Differential diagnosis ruled out infections, immune disorders, and Takayasu arteritis (TAK), a common cause of aortitis in children. Over a 1-year follow-up period, there was no recurrence and no stenotic lesions in large vessels. This finding suggests that the patient experienced transient LVV following COVID-19. Cytokine profile analysis performed before and after treatment revealed elevated levels of interleukin (IL)-6, IL-8, and IL-12/IL-23p40, typically associated with the active phase of TAK. Importantly, IL-17A and tumour necrosis factor-α levels were normal, as elevations in these cytokines have been linked to TAK recurrence. Notably, some cases of LVV following COVID-19 do not respond well to treatment; further research, including case accumulation and cytokine profile analysis, is needed to better predict prognosis.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815435","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":"Effective Use of Ultra-High Molecular Weight Polyethylene Cable and Krackow Suture for Stretched Out Patellar Tendon due to Scarring in a Case with Rheumatoid Arthritis Post-Total Knee Arthroplasty.","authors":"Eiji Kinoshita, Naoki Kondo, Osamu Tanifuji, Rika Kakutani, Nariaki Hao, Hiroyuki Kawashima","doi":"10.1093/mrcr/rxae074","DOIUrl":"https://doi.org/10.1093/mrcr/rxae074","url":null,"abstract":"<p><p>Patellar tendon rupture is a severe complication following total knee arthroplasty. We encountered a case of rheumatoid arthritis with an incomplete rupture of the patellar tendon post-total knee arthroplasty. An 84-year-old woman was diagnosed with an incomplete rupture of the right patellar tendon 3 months post-total knee arthroplasty of her right knee. The patient exhibited a 45° extension lag 6 months post-total knee arthroplasty, necessitating reconstruction surgery. Intraoperative findings revealed incomplete rupture of the patellar tendon that was stretched out, diagnosed as incomplete patellar tendon rupture. Due to knee valgus instability (passive knee valgus showed 20°), the thickness of the tibial insert was adjusted from 11 mm to 15 mm, resulting in improved valgus instability. The scarring region of the patellar tendon was resected to 10 mm in length, and the tendon was repaired using an ultra-high molecular weight polyethylene cable (Nesplon cable) and Krackow suture. The repair was secured by making a figure-8 pattern with the cable. After the reconstruction surgery, the knee was immobilized at 0° extension for 3 weeks, followed by the initiation of range-of-motion exercises. Three months later, the extension lag was reduced to -15°, and the patient could walk without orthosis and reported neither instability nor surgical site infection at 8 months after the surgery. In conclusion, this case is notable due to the rarity of incomplete (stretched out) patellar tendon rupture post-total knee arthroplasty and demonstrates the effectiveness of Nesplon cable with Krackow suture in reconstruction surgery.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815391","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}
Tokio Katakura, Tsuyoshi Shirai, Yusho Ishii, Hiroko Sato, Hiroshi Fujii
{"title":"Refractory skin ulcers and afebrile bacteremia with Staphylococcus aureus in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis: a case report.","authors":"Tokio Katakura, Tsuyoshi Shirai, Yusho Ishii, Hiroko Sato, Hiroshi Fujii","doi":"10.1093/mrcr/rxae082","DOIUrl":"https://doi.org/10.1093/mrcr/rxae082","url":null,"abstract":"<p><p>The skin ulcers sometimes appear in patients with anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive dermatomyositis (DM), and usually associates with disease activity. Here, we report a case of 41-year-old woman with anti-MDA5 antibody-positive DM, who developed refractory skin ulcers during the remission induction therapy, which were proven to be associated with clinically silent Staphylococcus aureus bacteremia with septic thrombi in her lung. The patient was referred to our hospital for the treatment of amyopathic DM with interstitial lung disease. Anti-MDA5-positive DM was diagnosed, and she was treated with triple therapy combined with tofacitinib because poor prognostic factors existed. Although the remission induction therapy improved most of symptoms, she developed erythematous rashes with ulcer on her left auricle and forearm, which were refractory to topical immunosuppressive medications. Despite the absence of systemic symptoms or elevated inflammatory markers, blood and wound cultures revealed S. aureus, and a new cavitary lesion was detected in her left lung. Subsequent antibiotic therapy resolved both the cutaneous and pulmonary lesions. This case highlights the importance of considering bacteremia and performing blood cultures when DM-related skin ulcers resist conventional treatments, even without fever during immunosuppressive therapy.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142815392","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":"Above-Knee Amputation in a 69-Year-Old Male with Gouty Tophi Complicated by Necrotizing Fasciitis - A Case Report.","authors":"Metasebia Worku Abebe, Abigya Aschalew Asfaw","doi":"10.1093/mrcr/rxae080","DOIUrl":"https://doi.org/10.1093/mrcr/rxae080","url":null,"abstract":"<p><p>Gout is the most prevalent form of inflammatory arthritis in males as a result of high uric acid levels that cause deposition of monosodium urate crystals in soft tissues, bones and joints. It commonly presents as swollen, erythematous and painful joint. Necrotizing fasciitis is rapidly progressing aggressive soft tissue infection that spreads along the fascial planes sparing the skin; it is very uncommon for necrotizing fasciitis to occur as a complication of gouty arthritis. We present here a case of a 69 year old male with known history of untreated gouty arthritis and tophi over the right foot that complicated into necrtotizing fasciits of the leg with extension to the upper thigh that was surgically debrided but also necessitated above knee amputation for infection control, survival and best possible functional outcome of the patient.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142808996","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":"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}