Modern Rheumatology Case Reports最新文献

{"title":"A case of aortoduodenal fistula caused by IgG4-related periaortitis.","authors":"Hideki Oka,&nbsp;Shuji Sumitomo,&nbsp;Hayato Shimizu,&nbsp;Maki Kanamori,&nbsp;Daisuke Yamashita,&nbsp;Hiroaki Nishioka,&nbsp;Koichiro Ohmura","doi":"10.1093/mrcr/rxac051","DOIUrl":"https://doi.org/10.1093/mrcr/rxac051","url":null,"abstract":"<p><p>An 86-year-old man who underwent endovascular aortic repair for impending rupture of an abdominal aortic aneurysm a year ago presented to our hospital because of fatigue and black stools. Multiple bacterial specimens were detected in blood cultures, and computed tomography following oral administration of gastrografin demonstrated gastrografin in the abdominal aorta. The diagnosis of aortic duodenal fistula was confirmed and emergency abdominal aortic replacement was performed. The pathological findings of the aorta included a large number of immunoglobulin G4 (IgG4)-positive plasma cells infiltrating all layers of the aortic wall, with particularly marked thickening of the adventitia. The serum IgG4 level was 241 mg/dl and IgG4-related periaortitis was diagnosed. Aortoduodenal fistula is a rare but fatal complication of IgG4-related periaortitis. Patients should be followed carefully after endovascular aortic repair for inflammatory abdominal aortic aneurysms.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"188-191"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10835462","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":"Fluorodeoxyglucose-positron emission tomography/computed tomography-positive ear lesions responsive to immunosuppressive therapy in a patient with otitis media with antineutrophil cytoplasmic antibody-associated vasculitis.","authors":"Yuki Murao,&nbsp;Yusuke Yoshida,&nbsp;Naoya Oka,&nbsp;Ai Yorishima,&nbsp;Sho Masuda,&nbsp;Tomohiro Sugimoto,&nbsp;Rina Ono,&nbsp;Yutaka Hirokawa,&nbsp;Shintaro Hirata","doi":"10.1093/mrcr/rxac044","DOIUrl":"https://doi.org/10.1093/mrcr/rxac044","url":null,"abstract":"<p><p>A 74-year-old woman presented with vertigo, left-beating nystagmus, and auditory disturbance 4 months prior, in whom a former physician suspected Meniere's disease. Her signs and symptoms mildly improved with a moderate dose of glucocorticoids, which was eventually tapered. Fluorodeoxyglucose (FDG)-positron emission tomography (PET)/computed tomography (CT) was performed 12 days prior to routine examination. Remarkable FDG uptake was observed in the surrounding areas of the bilateral Eustachian tubes and left middle ear, which was only partially detected on magnetic resonance imaging. The patient also tested positive for myeloperoxidase-antineutrophil cytoplasmic antibody (ANCA). She was admitted to our hospital and diagnosed with otitis media with ANCA-associated vasculitis (OMAAV) based on the classification criteria. Four months after immunosuppressive therapy, the abnormal ear findings were not observed on follow-up FDG-PET/CT. The clinical course of this case suggests that FDG-PET/CT can detect occult ear involvement better than do other modalities in patients with OMAAV. In addition, FDG-PET/CT-positive ear lesions responded to immunosuppressive therapy. Therefore, FDG-PET/CT can help distinguish OMAAV from other ear diseases with non-inflammatory aetiologies and detect occult treatment-responsive OMAAV lesions in the clinical setting.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"134-137"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10489186","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":"Mimic or coincidentally? TAFRO syndrome and systemic lupus erythematosus: A case-based review.","authors":"Burak Okyar,&nbsp;Bekir Torun,&nbsp;Esen Saba Öktem,&nbsp;Abdulkadir Yasir Bahar,&nbsp;Fatih Yıldız,&nbsp;Gözde Yıldırım Çetin","doi":"10.1093/mrcr/rxac045","DOIUrl":"https://doi.org/10.1093/mrcr/rxac045","url":null,"abstract":"<p><p>Castleman's disease (CD) is a rare, systemic disease with histopathological features of angiofollicular lymph node hyperplasia. In the literature, there are case-level reports that mimic or coexist with systemic lupus erythematosus (SLE) clinically and in the laboratory. Is this condition two separate diseases or is it an imitation of each other? A 73-year-old female patient was admitted to our clinic with arthritis, lymphadenopathy, fever, weight loss, and malar rash. He had a history of idiopathic thrombocytopenic purpura and thrombosis in the right leg tibialis posterior and dorsalis pedis arteries. Excisional lymphadenopathy biopsy indicated a diagnosis of hyaline-vascular-type CD. She had anti-nuclear antibody >1/80 homogeneous pattern, anti-double stranded DNA (anti-dsDNA), Anti-Smith (Sm) antibody positivity, hypocomplementemia (C3 and C4), pleural effusion, and pericardial effusion. For this reason, the classification criteria of the European League Against Rheumatism/American College of Rheumatology were studied. Clinical findings, idiopathic thrombocytopenic purpura history, antibody positivity, malar rash, and arthritis led us to the diagnosis of SLE. She was treated with 1 mg/kg/day prednisolone and hydroxychloroquine 200 mg 2 × 1. Azathioprine 2.5 mg/kg daily was added to the patient whose complaints did not improve. In the follow-ups, she completely recovered clinically and laboratory. SLE and CD are systemic diseases that overlap in many ways. The literature review shows that these two diseases may mimic each other or may coexist. This situation may be a reflection of a pathophysiological process that has not yet been clarified. This confusing process also affects the treatment decision. This confusing process also affects the treatment decision.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"271-275"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10489188","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":"Rheumatoid arthritis caused by non-tuberculous mycobacteria infection.","authors":"Hirokazu Takaoka,&nbsp;Taizo Shimomura,&nbsp;Hitoshi Suzushima","doi":"10.1093/mrcr/rxac001","DOIUrl":"https://doi.org/10.1093/mrcr/rxac001","url":null,"abstract":"<p><p>A 72-year-old Japanese woman had right digital flexor tenosynovitis with a non-tuberculous mycobacteria (NTM) infection, which was identified as Mycobacterium marinum in culture. She had been treated at another hospital with clarithromycin, rifampicin, and ethambutol for the non-tuberculous tenosynovitis. However, the swelling of her right hand worsened, and 5 months later, her left hand swelled and she exhibited symmetrical arthritis. Blood tests detected elevated serum C-reactive protein and rheumatoid factor positivity. Although rheumatoid arthritis (RA) was suspected and corticosteroid treatment was started, she came to our hospital because of the insufficient treatment effect. Musculoskeletal ultrasonography showed intra-articular and peritendinous power Doppler signal-positive symmetrical synovitis. A contrast-enhanced magnetic resonance imaging (MRI) evaluation of the left hand without NTM tenosynovitis revealed findings of inflammatory synovitis accompanied by bone marrow oedema. We diagnosed RA and started treatment with weekly low-dose methotrexate pulses and 2 weeks of tocilizumab administration; her symptoms then disappeared within 2 months. This is a rare case of RA manifested with NTM-associated arthritis.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"1-4"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10476087","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":"Arthritis remission with partial cartilage restoration in a 9-year-old female with hip chondrolysis following treatment with adalimumab.","authors":"Lampros Fotis,&nbsp;Anastasios Kanellopoulos,&nbsp;Olympia Papakonstantinou","doi":"10.1093/mrcr/rxac024","DOIUrl":"https://doi.org/10.1093/mrcr/rxac024","url":null,"abstract":"<p><p>Hip chondrolysis is observed primarily or secondary to other diseases and is a rare but yet debilitating disease, characterised by loss of cartilage of the femoral epiphysis and significant restriction of motion. We present the case of a 9-year-old female diagnosed with hip chondrolysis associated with probable juvenile psoriatic arthritis. Avoidance of weight-bearing activities and treatment with corticosteroids, methotrexate, and adalimumab followed by aquatic therapy resulted in clinical and radiographic improvement as well as partial cartilage regeneration.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"227-231"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10484971","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":"Lymphoid follicular hyperplasia in patients with systemic lupus erythematosus after multiple cycles of rituximab.","authors":"Ingrid Ruiz-Ordóñez,&nbsp;Víctor A Santos,&nbsp;Fabio Bonilla-Abadía,&nbsp;Nhora Silva,&nbsp;Aura Sánchez,&nbsp;Gabriel J Tobón,&nbsp;Carlos A Cañas","doi":"10.1093/mrcr/rxac066","DOIUrl":"10.1093/mrcr/rxac066","url":null,"abstract":"<p><p>Rituximab is indicated in some patients with refractory systemic lupus erythematosus (SLE). Occasionally, this medication is required in chronic form to maintain control of the disease. We described two patients who developed lymphoid follicular hyperplasia (LFH) after multiple cycles of rituximab and evaluated the expression of B cell activating factor belonging to the tumor necrosis factor (TNF) family (BAFF) and its receptors [BAFF-receptor (BAFF-R) and B cell maturation antigen (BCMA)], as possible factors related to lymphoid node enlargement. Two patients with SLE completed six and nine cycles of rituximab (1 g every 2 weeks) indicated each 9 months, achieving remission for 5 and 7 years, respectively, when developed prominent lymphadenopathies. Biopsies showed LFH. Haematological neoplasms were ruled out. Immunohistochemistry showed BAFF overexpression in the follicles, and moderate expression of BAFF-R confined to the mantle zone and BCMA to the germinal centre. Belimumab B cell activating factor belonging to the TNF family (anti-BAFF therapy) was started with positive effects on the clinical condition. LFH can develop in patients with SLE who received multiple cycles of rituximab. BAFF overexpression and moderate expression of BAFF-R and BCMA in lymph nodes were seen. These findings added to the improvement with the change to belimumab could suggest that LFH after cluster of differentiation (CD20) depletion therapy may be associated with a compensatory overexpression of BAFF and its receptors.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"78-81"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10486165","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":"Four cases of dermatomyositis with abnormally high anti-MDA-5 antibody titres and not high levels of serum ferritin.","authors":"Daichi Umemoto,&nbsp;Shuji Sumitomo,&nbsp;Shohei Fujita,&nbsp;Hayato Shimizu,&nbsp;Hideki Oka,&nbsp;Maki Kanamori,&nbsp;Hiroaki Nishioka,&nbsp;Koichiro Ohmura","doi":"10.1093/mrcr/rxac053","DOIUrl":"https://doi.org/10.1093/mrcr/rxac053","url":null,"abstract":"<p><p>Anti-melanoma differentiation-associated gene 5 (MDA-5) antibody-positive dermatomyositis is a fatal disease presenting with rapidly progressive interstitial lung disease. High ferritin levels are a well-known poor prognostic factor. A high anti-MDA-5 antibody titre was also recently identified as a poor prognostic factor. We encountered four cases that had extremely high anti-MDA-5 antibody titres without high levels of ferritin in the initial examination. All cases were female with ages ranging between 29 and 54 years (mean age, 44 years). In the initial examination, anti-MDA-5 antibody titres were 2060-3040 (normal range, <32 index), ferritin levels were 87-480 ng/ml (normal range, 2.6-129.4 ng/ml), KL-6 level was 186-1806 U/ml (normal range, <500 U/ml), and creatine kinase level was normal in all patients. One patient had respiratory distress on exertion. Computed Tomography (CT) images showed mild ground-glass attenuation/reticular shadows near the pleura in all patients. Three patients were treated with a combination of high-dose glucocorticoids, intermittent intravenous cyclophosphamide, and calcineurin inhibitors, and two required plasma exchange due to the worsening of lung lesion. In these patients, ferritin and KL-6 levels tended to elevate after the beginning of treatment. Very mild pulmonary lesions disappeared in one patient treated with moderate doses of a glucocorticoid and calcineurin inhibitor. All patients survived, and one required oxygen on exertion at discharge. The condition of patients with abnormally high anti-MDA-5 antibody titres may deteriorate even though ferritin levels were not high and lung shadows are minimal at presentation. Therefore, intensive treatment needs to be considered early in the course of the disease regardless of the serum ferritin level.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"96-101"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10498574","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":"Bell's palsy with facial bone involvement: A rare presentation of chronic nonbacterial osteomyelitis with literature review.","authors":"Haider A Ailumerab,&nbsp;Cassyanne L Aguiar","doi":"10.1093/mrcr/rxac058","DOIUrl":"https://doi.org/10.1093/mrcr/rxac058","url":null,"abstract":"<p><p>Chronic nonbacterial osteomyelitis (CNO) is a chronic, sterile, inflammatory disease. It primarily presents with nonspecific bone pain and swelling but ultimately can cause bone destruction and deformities, if left untreated. The involvement of the cranial bones (apart from the mandible) is rare in CNO. In this report, we present a rare case of CNO affecting facial and cranial bones presenting as facial palsy with a review of the literature about similar affection. A 10-year-old, previously healthy female was initially evaluated for swelling of the left side of her face with slight tenderness on palpation, but no fever. Her complete blood count was unremarkable, her inflammatory markers were elevated (C-reactive protein 7.5 mg/dl and erythrocyte sedimentation rate 104 mm/h), and CT of facial and skull bones and MRI of brain showed a destructive osseous process involving the left maxillary, zygomatic, sphenoid bones and the clivus. Bone biopsy of the left maxilla showed fibrous dysplasia with abscess formation, most consistent with an infectious aetiology (acute osteomyelitis). She was started on oral clindamycin for a 3-month course. The facial swelling improved after starting clindamycin, but on her sixth week of treatment, she developed right-sided Bell's palsy. An MRI of the brain showed hyperenhancement of the right seventh cranial nerve. A month later, she was evaluated for right wrist and knee swelling, pain, and limitation of movement. Skeletal survey and MRI showed multifocal lesions with mixed sclerosis and lucency. Her inflammatory markers continued to be elevated. Another bone biopsy of the right radius showed similar findings of destruction with no evidence of malignancy. She was ultimately diagnosed with CNO. She was started on nonsteroidal anti-inflammatory drugs with gastric protection and regular follow-up. Over more than a year of follow-up, the patient's inflammatory markers remain normal, and joint swelling/limitation has remained in remission. We found five additional cases in the literature that presented with a similar presentation. To our knowledge, our patient is the first reported case in the USA involving the cranial/facial bones apart from the mandible presenting with facial palsy. The affection of the facial bones (apart from the mandible) in CNO is very rare, but the awareness of such a presentation by the clinician is an important aspect of reaching the diagnosis.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"298-301"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10485955","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":"Azathioprine-induced hypersensitivity reaction mimicking sepsis in a patient with systemic lupus erythematosus.","authors":"Paige L McKenzie,&nbsp;Yusuf Chao,&nbsp;Sapna Pathak,&nbsp;Salahuddin Kazi","doi":"10.1093/mrcr/rxac061","DOIUrl":"https://doi.org/10.1093/mrcr/rxac061","url":null,"abstract":"<p><p>A 21-year-old woman with a history of systemic lupus erythematosus presented to the emergency department with acute-onset nausea, vomiting, and fevers. Two weeks prior, she was started on azathioprine 50 mg daily by her outpatient rheumatologist; the dose was up-titrated to 100 mg when repeat blood work showed no drug toxicity. The morning after increasing her dose, she was awoken by recurrent emesis. At presentation, she was febrile, tachycardic, and hypotensive. Her exam showed mild, generalised abdominal tenderness but was otherwise unremarkable. Lab work demonstrated elevated inflammatory markers, elevated liver transaminases, and stable hypocomplementemia. Chest X-ray and computed tomography abdomen/pelvis were unrevealing. She was given intravenous fluids and broad-spectrum antibiotics, and azathioprine was held. A thorough infectious workup returned negative. A flare of her systemic lupus erythematosus was considered but deemed an unlikely explanation of her systemic inflammatory response syndrome. With azathioprine discontinuation, she made a rapid, near-complete recovery within 24 h of admission, suggesting a diagnosis of azathioprine hypersensitivity syndrome. This case exemplifies the difficulty in distinguishing azathioprine hypersensitivity from mimickers such as infection and underlying autoimmune disease flare. Prompt recognition of hypersensitivity can lead to appropriate discontinuation of the drug and prevent future morbidity.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"74-77"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10486160","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":"Intracranial vascular stenosis in giant cell arteritis successfully treated by two balloon angioplasty procedures.","authors":"Kensaku Takatsuki,&nbsp;Yuki Kojima,&nbsp;Yasuhito Ikeuchi,&nbsp;Jiro Kitayama,&nbsp;Atsushi Tanaka,&nbsp;Yasushi Inoue","doi":"10.1093/mrcr/rxac080","DOIUrl":"https://doi.org/10.1093/mrcr/rxac080","url":null,"abstract":"<p><p>Intracranial vascular stenosis is rarely associated with giant cell arteritis (GCA), and the prognosis for stroke caused by GCA is poor. Despite its unfavourable outcome, the strategy to manage this involvement and the indication of endovascular treatment are not well defined in the latest guidelines or recommendations. Here, we present a case in a 68-year-old woman, which was refractory to medical therapy, but successfully treated by two balloon angioplasty procedures. She was admitted to our department with lower extremity stiffness and left visual disturbance. GCA was clinically diagnosed by the wall thickening of the temporal artery and the aorta. Hemiparesis and motor aphasia developed shortly after intravenous methylprednisolone pulse therapy, and magnetic resonance imaging revealed acute cerebral infarction with severe stenosis at the end of the left internal carotid artery. Balloon angioplasty was tried initially with improvement in her symptoms and additionally performed to treat restenosis without any significant adverse events. Her symptoms markedly improved with no recurrence until 8 months after discharge. We also review 10 similar cases reported in the literature. Although further evidence is needed to confirm the usefulness and safety of balloon angioplasty for intracranial GCA, this case report provides valuable information about the endovascular therapy for GCA.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 1","pages":"166-171"},"PeriodicalIF":0.0,"publicationDate":"2023-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10486986","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}