{"title":"Strongyloidiasis presenting during treatment of low-dose glucocorticoid plus rituximab for anti-signal recognition particle antibody-positive polymyositis: A case report.","authors":"Ryo Oshima, Takaharu Matsuhisa, Takeshi Kondo, Motoki Sato, Juichi Sato, Masahiro Yamamura","doi":"10.1093/mrcr/rxac083","DOIUrl":"https://doi.org/10.1093/mrcr/rxac083","url":null,"abstract":"<p><p>Strongyloidiasis, an intestinal parasitic infection caused by Strongyloides stercoralis, rarely occurs in Japan. When treated with immunosuppressive drugs, two potentially lethal conditions, hyperinfection and dissemination, may develop in asymptomatic carriers of this parasite. We report the development of strongyloidiasis during treatment of polymyositis with glucocorticoids plus rituximab (RTX). A 44-year-old woman had been diagnosed with anti-signal recognition particle antibody-positive polymyositis with interstitial pneumonia 6 years previously, for which she had recently been receiving prednisolone at 5 mg/day and RTX at 375 mg/m2 twice every 3 months. Her condition appeared to be well controlled. She was admitted to our hospital with a 1-month history of chronic diarrhoea and epigastric pain. Standard microscopic examination of a sample of faeces revealed the presence of S. stercoralis; however, serologic testing for parasites was negative. Treatment with ivermectin alleviated her inflammatory diarrhoea and eradicated the faecal parasites. We believe that our patient had an exacerbation of S. stercoralis infection (hyperinfection syndrome) that was exacerbated by low-dose glucocorticoids plus RTX. Strongyloidiasis should be considered in immunocompromised individuals with unexplained diarrhoea, even in non-endemic areas.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"113-116"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10487424","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 of septic arthritis of the elbow joint in rheumatoid arthritis diagnosed by arthroscopic synovectomy.","authors":"Masako Hayashibara, Hiroshi Hagino, Ikuta Hayashi, Keita Nagira, Yuta Takasu, Daichi Mukunoki, Nagashima Hideki","doi":"10.1093/mrcr/rxac048","DOIUrl":"https://doi.org/10.1093/mrcr/rxac048","url":null,"abstract":"<p><p>We report a case of methicillin-resistant Staphylococcus aureus (MRSA) septic arthritis of the elbow detected by arthroscopic synovectomy in an 81-year-old woman with rheumatoid arthritis (RA) who was initially diagnosed with a rheumatoid arthritis flare-up. The patient was administered abatacept, an antirheumatic biological agent, as the synovial fluid culture was negative. Destruction of the joint progressed despite medication, and the patient underwent arthroscopic synovectomy. MRSA was detected in the culture of the synovium that was collected intraoperatively, and septic arthritis was diagnosed. The infection subsided with anti-MRSA antibiotics, but the patient continued to experience moderate pain and limited motion. In RA patients, it might be difficult to differentiate minor findings from infection. Arthroscopic synovectomy is one of the selectable procedures that should be actively considered when infection is suspected.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"24-27"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10489662","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":"Appearance of anti-MDA5 antibody-positive dermatomyositis after COVID-19 vaccination.","authors":"Tomohiro Sugimoto, Ai Yorishima, Naoya Oka, Sho Masuda, Naoki Nakamoto, Genki Kidoguchi, Hirofumi Watanabe, Yusuke Yoshida, Sho Mokuda, Shintaro Hirata","doi":"10.1093/mrcr/rxac064","DOIUrl":"https://doi.org/10.1093/mrcr/rxac064","url":null,"abstract":"<p><p>The direct causes of dermatomyositis, a common autoimmune disease, have not yet been accurately identified, but several studies have linked this condition to various patient-associated and environmental factors, such as viral infections and area of residence. In the present report, we describe our experience with a patient presenting with anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive dermatomyositis, which developed after vaccination against coronavirus disease 2019 (COVID-19). This patient was simultaneously diagnosed with anti-glutamic acid decarboxylase antibody-positive slowly progressive insulin-dependent diabetes (SPIDDM); her human leucocyte antigen test revealed that she expressed the DRB1*04:05 allele. This is important as this genotype is known to increase susceptibility to both anti-MDA5 antibody-positive dermatomyositis and type I diabetes. To the best of our knowledge, this is the first case of dermatomyositis complicated by SPIDDM identified after COVID-19 vaccination against COVID-19 and presenting with an underlying susceptible genotype. The patient's genetic predisposition may also be important for the development of autoimmune disease after COVID-19 vaccination.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"108-112"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10503773","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":"Epstein-Barr virus-associated haemophagocytic lymphohistiocytosis complicated by neurological involvement in a patient with dermatomyositis: A case report and literature review.","authors":"Lijuan Xiong, Tingting Xu, Liang Tang","doi":"10.1093/mrcr/rxac056","DOIUrl":"https://doi.org/10.1093/mrcr/rxac056","url":null,"abstract":"<p><p>Haemophagocytic lymphohistiocytosis (HLH) is an extremely high mortality condition that can occur at any age and lacks specific diagnostic characteristics. Despite its rarity, HLH is increasingly alarming because of the high mortality rate. It is a systemic hyperinflammatory immune response syndrome associated with abnormal activation of T cells and macrophages, which may be caused by genetic mutations or acquired factors, such as infection, autoimmune condition, and malignancy. Here, we present a fatal case of Epstein-Barr virus-associated HLH complicated by neurological involvement in a patient with dermatomyositis. We describe and discuss the pathological features, classification, and effective treatment options of HLH and discuss separately the special characteristics of Epstein-Barr virus-associated HLH and dermatomyositis-associated HLH.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"293-297"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10542180","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":"Visceral disseminated varicella zoster virus infection during non-intensive maintenance therapy in a patient with systemic lupus erythematosus.","authors":"Koto Hattori, Shigeru Tanaka, Junichi Ishikawa, Yoko Yabe, Taro Iwamoto, Shunsuke Furuta, Kei Ikeda, Kotaro Suzuki, Hiroshi Nakajima","doi":"10.1093/mrcr/rxac054","DOIUrl":"https://doi.org/10.1093/mrcr/rxac054","url":null,"abstract":"<p><p>Visceral disseminated varicella zoster virus infection (VD-VZV) is a rare complication in immunocompromised patients. Although systemic lupus erythematosus (SLE) patients have a higher risk of VZV infection, only a few reports describe VD-VZV in SLE. Here, we report a 48-year-old woman with SLE who had received maintenance therapy. She was transferred to the hospital because of severe epigastric pain. There were no significant abnormalities in abdominal computed tomography and upper gastrointestinal endoscopy. On hospital day 4, she developed vesicular eruption on her face and abdomen. VZV antigen was detected in specimens obtained from skin lesions, and treatment with acyclovir was started. VZV DNA in blood turned out to be positive, and the epigastric pain was thought to be caused by VD-VZV. There is a risk of VD-VZV in patients with SLE, even in those receiving non-intensive maintenance therapy.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"57-59"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10835483","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}
Sebastian Molina Rios, Rigoberto Rojas Martinez, Gloria Marcela Estévez Ramirez, Yimy Francisco Medina
{"title":"Systemic lupus erythematosus and antiphospholipid syndrome after COVID-19 vaccination. A case report.","authors":"Sebastian Molina Rios, Rigoberto Rojas Martinez, Gloria Marcela Estévez Ramirez, Yimy Francisco Medina","doi":"10.1093/mrcr/rxac018","DOIUrl":"https://doi.org/10.1093/mrcr/rxac018","url":null,"abstract":"<p><p>Coronavirus disease 2019 (COVID-19) vaccines have some adverse effects, mostly mild. However, by presenting an immunological challenge to the individual, they could infrequently trigger immune-mediated diseases (IMDs). We report the case of a 42-year-old woman, with no previous medical history, who received the first dose of vaccine against COVID-19 and developed inflammatory arthralgias, associated with sudden-onset dyspnoea and hypoxemia. Pulmonary thromboembolism was documented, and the diagnosis of systemic lupus erythematosus (SLE) and secondary antiphospholipid syndrome (APS) was suspected. Autoantibodies were measured confirming this suspicion. After a few days, she presented a massive pericardial effusion with cardiac tamponade that required surgical management. Treatment with azathioprine, hydroxychloroquine, corticosteroids, and anticoagulation was indicated with improvement of all her symptoms. There is controversy regarding the potential of COVID-19 vaccines to induce autoimmunity. Studies addressing the safety of using these vaccines have reported the occurrence of mild local and systemic reactions, most frequently in young adults. So far, there are few reports of patients who have developed autoimmune or autoinflammatory diseases after getting vaccinated with any of the COVID-19 vaccines. To the best of our knowledge, to date, this is one of the first cases of new-onset SLE and secondary APS after COVID-19 vaccination.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"43-46"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8903504/pdf/rxac018.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10850946","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Short stature as an initial presenting presentation of unicentric Castleman disease in a child: A case report with long-term follow-up and a literature review.","authors":"Kaori Sonoda, Utako Kaneko, Makoto Hiura, Yoshiaki Kinoshita, Hajime Umezu, Shuichi Ito, Akihiko Saitoh, Chihaya Imai","doi":"10.1093/mrcr/rxac034","DOIUrl":"https://doi.org/10.1093/mrcr/rxac034","url":null,"abstract":"<p><p>Short stature is a common clinical condition in paediatric outpatient clinics and is associated with various clinical conditions, ranging from normal variants to severe diseases. Short stature is known to be caused by chronic inflammatory conditions, in which over-produced inflammatory cytokines are reported to be involved in growth suppression. Castleman disease is a rare lymphoproliferative disorder known as a chronic inflammatory disease with overproduction of interleukin 6, which often causes systemic symptoms such as fever, fatigue, weight loss, and night sweats. Here, we report the case of a 10-year-old female diagnosed with unicentric Castleman disease, who presented with short stature as the sole clinical sign but lacked typical systemic symptoms of Castleman disease. An elevated serum C-reactive protein level led us to suspect a chronic inflammatory condition, and we found an intra-abdominal tumour that was histopathologically confirmed as Castleman disease. The tumour removal resulted in a steady catch-up in her height in the six years following the surgery. We also present a brief review of relevant literature on paediatric cases of Castleman disease associated with growth impairment. Clinicians should be aware that chronic inflammatory conditions can cause growth impairment, which may be a key clinical manifestation of such conditions.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"261-266"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10484693","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":"Multisystem inflammatory syndrome in children (MIS-C) without antecedent clinical symptoms related to COVID-19: The increasing importance of distinguishing MIS-C in patients with Kawasaki disease-like features.","authors":"Masato Ozaki, Tomohiro Hori, Yuki Miwa, Takahiro Yamamoto, Kazuo Kubota, Minako Kawamoto, Norio Kawamoto, Hidenori Ohnishi","doi":"10.1093/mrcr/rxac008","DOIUrl":"10.1093/mrcr/rxac008","url":null,"abstract":"Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infections in most children result in an asymptomatic or milder course of coronavirus disease 2019 (COVID-19) compared with infections in adults [1]. How-ever, SARS-CoV-2 infection in children may rarely result in hyperinflammatory features similar to Kawasaki disease (KD) and toxic shock syndrome, known as ‘multisystem inflammatory syndrome in children’ (MIS-C). We report a Japanese case of MIS-C without antecedent clinical symptoms related to COVID-19.A9-year-old Japanese boy presented to our hospital because he had a fever 3 days prior to his visit, after which he developed conjunctival hyperaemia, redness of the lips (Figure 1(a)), and diarrhoea. He was not vaccinated against SARS-CoV-2, and he had no apparent history of illness and no respiratory symptoms or fever suggestive of COVID-19 within the preceding few months. He had not knowingly had close contact","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"202-204"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9383363/pdf/rxac008.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10484938","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"A case of giant cell arteritis complicated by polymyalgia rheumatica with scalp necrosis and refractory jaw claudication.","authors":"Shumpei Kosaka, Masao Nawata, Kenji Yamazumi, Aya Nawata, Katsumi Nakamura, Kazuyoshi Saito, Yoshiya Tanaka","doi":"10.1093/mrcr/rxac084","DOIUrl":"https://doi.org/10.1093/mrcr/rxac084","url":null,"abstract":"<p><p>Scalp necrosis is a rare complication of giant cell arteritis (GCA); however, it is a predictor of severe disease. In this case study, a patient presented with GCA complicated by polymyalgia rheumatica with scalp necrosis. An 86-year-old woman was admitted to the hospital for pulsating headache, scalp pain, jaw claudication, and generalised pain. Bilateral temporal arteries were found to be distended and pulseless, and scalp necrosis was observed in the parietal region. Simultaneous high-resolution contrast-enhanced magnetic resonance imaging (MRI) sequences of the head, shoulder, and hip showed staining around the bilateral shallow temporal arteries, shoulder, and hip joints, which was confirmed as GCA with polymyalgia rheumatica using other examination findings. After treatment with early induction remission therapy, scalp necrosis healed, but jaw claudication persisted. Six months after the start of treatment, scalp necrosis was cured to full hair growth. Despite remission induction therapy combined with tocilizumab, the patient had persistent jaw claudication for several months. At that time, a high-resolution contrast-enhanced MRI re-examination was useful in assessing disease activity. GCA with scalp necrosis may cause prolonged jaw claudication reflecting the progression of ischaemic lesions, whereas the disease activity can be accurately assessed by combining MRI studies.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"177-181"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10485545","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 of glucocorticoid-resistant adult Still's disease complicated by pulmonary hypertension and interstitial lung disease.","authors":"Katsuhide Kusaka, Ippei Miyagawa, Shunpei Kosaka, Satsuki Matsunaga, Shingo Nakayamada, Yoshiya Tanaka","doi":"10.1093/mrcr/rxac081","DOIUrl":"https://doi.org/10.1093/mrcr/rxac081","url":null,"abstract":"<p><p>Adult Still's disease (ASD) is rarely complicated by pulmonary hypertension (PH). A 76-year-old woman experienced ASD relapse with repeated exacerbation of PH and interstitial lung disease. Although she had been treated with immunosuppressive agents and pulmonary vasodilators, the ASD relapsed with fever, rash, increased inflammatory responses and exacerbated interstitial lung disease, and PH. The pathology of PH appeared to encompass groups 1 [pulmonary arterial hypertension (PAH)], 1' [pulmonary veno-occlusive disease (PVOD)], and 3. Remission induction therapy with high-dose glucocorticoid and tocilizumab was administered, and switching or adding pulmonary vasodilators was also attempted. As the disease activity of ASD decreased, the mean pulmonary arterial pressure and pulmonary vascular resistance improved. PH is an extremely rare form of organ dysfunction in individuals with ASD. Like other systemic autoimmune diseases, PH (PAH or PVOD) can determine the prognosis of ASD. Because of PH's rarity, it is important to sufficiently evaluate its pathology, considering the possibility that PH is not clinically classified as PAH (group 1), and to administer immunosuppressive therapy and vasodilators according to the pathology.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"182-187"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10486987","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}