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Antisynthetase Syndrome in a Patient with Pulmonary Embolism and Nonbacterial Thrombotic Endocarditis. 肺栓塞合并非细菌性血栓性心内膜炎患者的抗合成酶综合征。
Pub Date : 2023-01-01 DOI: 10.1155/2023/9068597
Anusha Vege, Jesse Beery, Areeba Kara

Antisynthetase syndrome is a rare autoimmune disease within the subset of idiopathic inflammatory myopathies. The diagnostic criteria include the presence of an aminoacyl-tRNA synthetase antibody, and typical clinical findings, including myositis, mechanic's hands, Raynaud phenomenon, unexplained fever, and interstitial lung disease. We describe a case of a 59-year-old male who presented with a 1-month history of progressive purplish discoloration and pain of the fingertips, dyspnea, cough, weight loss, fatigue, and who developed progressive proximal muscle weakness and dysphagia. Investigations revealed pulmonic valve and mitral valve marantic endocarditis, pulmonary embolism, myositis, organizing pneumonia, and elevation of anti-OJ antibodies. He was diagnosed with antisynthetase syndrome and treated with high dose corticosteroids and mycophenolate mofetil with a fair response.

抗合成酶综合征是一种罕见的自身免疫性疾病,属于特发性炎性肌病的子集。诊断标准包括存在氨基酰- trna合成酶抗体,以及典型的临床表现,包括肌炎、机械性手、雷诺现象、不明原因发热和间质性肺疾病。我们描述了一个59岁的男性病例,他表现为1个月的进行性紫色变和指尖疼痛,呼吸困难,咳嗽,体重减轻,疲劳,并发展为进行性近端肌肉无力和吞咽困难。调查显示肺动脉瓣和二尖瓣血管性心内膜炎、肺栓塞、肌炎、组织性肺炎和抗oj抗体升高。他被诊断为抗合成酶综合征,并接受大剂量皮质类固醇和霉酚酸酯治疗,反应良好。
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引用次数: 2
Peripheral Spondyloarthritis Presenting with Fever and Severe Systemic Inflammatory Response Mimicking Infection: A Case Series and Literature Review. 周围性脊柱炎表现为发热和严重的全身炎症反应模拟感染:一个病例系列和文献回顾。
Pub Date : 2023-01-01 DOI: 10.1155/2023/6651961
Ibrahim Abdulmomen, Eman Satti, Basem Awadh

Objective: To describe four peripheral spondyloarthritis patients presenting with fever and severe systemic inflammatory response mimicking infection.

Methods: Between 2017 and 2019, four patients with the final diagnosis of peripheral spondyloarthritis had atypical presentation of fever and severe systemic inflammatory response requiring hospital admission and extensive workup.

Results: We reported four patients who were admitted to the hospital for fever and arthritis. They all had laboratory tests of the severe systemic inflammatory response (leukocytosis, thrombocytosis, high ESR, and high CRP) concerning infection. They underwent extensive workup for infectious causes, including septic arthritis, which came back negative. Other rheumatic diseases that are known to present with fever such as adult-onset Still's disease, reactive arthritis, and crystal arthritis were all excluded. The final diagnosis of spondyloarthritis was made during their follow-up: three patients with peripheral spondyloarthritis and one with psoriatic arthritis. All patients received conventional DMARDs (methotrexate and sulfasalazine) and two patients received tumor necrosis factor inhibitors in addition to conventional DMARDs to control their disease.

Conclusion: We observed a subgroup of peripheral spondyloarthritis patients presenting with fever and severe systemic inflammatory response requiring hospitalization. Recognition of this subgroup is important and should be considered once an infection is ruled out.

目的:描述4例以发热和严重的全身炎症反应模拟感染为表现的周围性脊柱炎患者。方法:2017年至2019年,4例最终诊断为周围性脊柱炎的患者出现不典型的发热和严重的全身炎症反应,需要住院和广泛的随访。结果:我们报告了4例因发烧和关节炎住院的患者。他们都进行了与感染有关的严重全身炎症反应(白细胞增多、血小板增多、高ESR和高CRP)的实验室检查。他们接受了广泛的传染病检查,包括感染性关节炎,结果呈阴性。其他已知以发热为表现的风湿病,如成人发病的斯蒂尔氏病、反应性关节炎和水晶关节炎均被排除在外。脊柱关节炎的最终诊断是在随访期间做出的:三名患者患有外周性脊柱关节炎,一名患者患有银屑病关节炎。所有患者均接受常规DMARDs(甲氨蝶呤和柳氮磺胺吡啶)治疗,2例患者在常规DMARDs治疗的基础上接受肿瘤坏死因子抑制剂治疗以控制病情。结论:我们观察到一个亚组的周围性脊柱炎患者表现为发烧和严重的全身炎症反应,需要住院治疗。识别这一亚群是很重要的,一旦排除感染就应予以考虑。
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引用次数: 0
Immune-Mediated Necrotizing Myopathy Manifesting after Five Years of Statin Therapy. 他汀类药物治疗5年后出现的免疫介导的坏死性肌病。
Pub Date : 2023-01-01 DOI: 10.1155/2023/1178035
Nathan G DeRon, Francis Fischer, Dylan Lopez, Elizabeth C Brewer

Immune-mediated necrotizing myopathy (IMNM) is an increasingly common and serious condition in which autoantibodies attack muscle fibers causing clinically significant muscle weakness, fatigue, and myalgias. Recognizing the clinical presentation of IMNM is difficult but necessary, as rapid intervention decreases morbidity. We present a case of a 53-year-old female with IMNM induced by statin therapy with confirmed anti-3-hydroxy-3-methylglutaryl coenzyme A reductase antibodies present on serologic testing. The patient's statin therapy was halted, and the patient was provided with one dose of methylprednisolone and ongoing therapy with mycophenolate. She showed subsequent slow improvements in her muscle weakness and myalgias. It is important for clinicians to be aware of the possible consequences of statin therapy, as these drugs are generally regarded as benign in the medical community. Clinicians should also be aware that statin-induced myopathy can occur at any time during statin therapy. The condition does not necessarily correlate with beginning a new statin medication, as demonstrated in this case in which the patient was on chronic statin therapy before developing symptoms. Continued clinician education and building the fund of medical knowledge regarding this disease are vital to enable clinicians to recognize this disease and act promptly to reduce patient morbidity and improve outcomes.

免疫介导的坏死性肌病(IMNM)是一种越来越常见和严重的疾病,其自身抗体攻击肌肉纤维,导致临床显着的肌肉无力,疲劳和肌痛。认识IMNM的临床表现是困难的,但必要的,因为快速干预可以降低发病率。我们报告一例53岁的女性,他汀类药物治疗引起IMNM,血清学检测证实抗3-羟基-3-甲基戊二酰辅酶a还原酶抗体存在。患者停止他汀类药物治疗,给予患者一剂甲基强的松龙和持续的霉酚酸盐治疗。随后,她的肌肉无力和肌痛症状缓慢改善。对于临床医生来说,重要的是要意识到他汀类药物治疗可能产生的后果,因为这些药物在医学界通常被认为是良性的。临床医生也应该意识到他汀类药物诱导的肌病可能在他汀类药物治疗期间的任何时候发生。这种情况并不一定与开始新的他汀类药物有关,正如本病例所证明的那样,患者在出现症状之前一直在接受慢性他汀类药物治疗。持续的临床医生教育和建立关于这种疾病的医学知识基金对于使临床医生认识到这种疾病并及时采取行动以减少患者发病率和改善结果至关重要。
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引用次数: 0
A Case of Previously Undiagnosed Systemic Lupus Erythematosus and Mycobacterium tuberculosis Infection Presenting as Diffuse Alveolar Hemorrhage. 以弥漫性肺泡出血为表现的系统性红斑狼疮合并结核分枝杆菌感染1例。
Pub Date : 2023-01-01 DOI: 10.1155/2023/3686772
Henna Iqbal, Benny Screws, Muhammad S Khan

Diffuse alveolar hemorrhage (DAH) is described as the collection of blood in alveolar spaces caused by damaged pulmonary vasculature. It often presents as a life-threatening medical emergency that requires urgent medical intervention along with timely diagnosis and management of the underlying cause. We hereby report a 19-year-old female who presented with clinical and radiological characteristics consistent with DAH. Laboratory workup studies revealed a diagnosis of systemic lupus erythematosus (SLE) as well as Mycobacterium tuberculosis (MTB) infection. This report describes an extremely unusual case of undiagnosed SLE and coexistent tuberculosis presenting as DAH. This leads to an interesting possibility of risks in patients with immune-mediated vasculitis towards developing severe pulmonary disease in the setting of pulmonary mycobacterial infection.

弥漫性肺泡出血(DAH)被描述为肺血管受损引起的肺泡间隙血液聚集。它通常表现为危及生命的医疗紧急情况,需要紧急医疗干预以及及时诊断和处理根本原因。我们在此报告一位19岁的女性,她的临床和放射学特征与DAH一致。实验室检查显示诊断为系统性红斑狼疮(SLE)以及结核分枝杆菌(MTB)感染。本报告描述了一个极其罕见的病例,未确诊的SLE和并发肺结核,表现为DAH。这导致了一种有趣的可能性,即在肺分枝杆菌感染的情况下,免疫介导的血管炎患者发生严重肺部疾病的风险。
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引用次数: 1
Childhood-Onset COPA Syndrome Recognized Retrospectively in the Context of Polyarticular Juvenile Idiopathic Arthritis and Rheumatoid Arthritis. 儿童期发作的COPA综合征在多关节幼年特发性关节炎和类风湿关节炎的背景下回顾性识别。
Pub Date : 2023-01-01 DOI: 10.1155/2023/3240245
Roko P A Nikolic, Cristina Moran Toro

COPA syndrome is a very rare autoinflammatory disorder manifesting with childhood-onset arthritis and pulmonary and renal disease, of which awareness may remain lacking. We present the case of a twenty-year-old male patient seen in the Young Adults with Rheumatic Disease clinic. Initially diagnosed with seropositive polyarticular juvenile idiopathic arthritis, the patient's early childhood complaints of fatiguability, paroxysmal dyspnea, and pneumonia-like episodes were long to be felt unrelated to his arthritis. Upon transition to adult rheumatology care, a thorough review of the patient's history prompted imaging which revealed interstitial lung disease. Restrictive spirometry and genetic testing confirmed the retrospective diagnosis of COPA syndrome.

COPA综合征是一种非常罕见的自身炎症性疾病,表现为儿童期发病的关节炎、肺部和肾脏疾病,对这种疾病的认识可能仍然缺乏。我们提出的情况下,一个二十岁的男性患者看到的年轻成人风湿病诊所。最初诊断为血清阳性多关节幼年特发性关节炎,患者童年早期的疲劳、阵发性呼吸困难和肺炎样发作,长期被认为与他的关节炎无关。在过渡到成人风湿病护理,彻底检讨病人的历史提示影像学显示间质性肺疾病。限制性肺活量测定和基因检测证实回顾性诊断为COPA综合征。
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引用次数: 0
Systemic Mastocytosis: A Mimicker of Reactive Arthritis. 系统性肥大细胞增多症:反应性关节炎的模拟者。
Pub Date : 2023-01-01 DOI: 10.1155/2023/6655005
Oussama G Nasrallah, Razan Mohty, Jean El-Cheikh, Mira Merashli

Objectives: Illustration of a case of systemic mastocytosis mimicking reactive arthritis in the absence of an infectious etiology.

Methods: Review of the patient's medical records.

Results: We report a case of systemic mastocytosis relapse, presenting with pancytopenia accompanied by knee monoarthritis, cystitis, and bilateral conjunctivitis occurring simultaneously at the same time interval within 2-4 days, mimicking reactive arthritis in the absence of an infectious etiology.

Conclusion: Our case demonstrated reactive arthritis features (triad of urethritis, conjunctivitis, and arthritis) without an infectious trigger but rather a relapse of mastocytosis. We should think outside the box when faced with such a clinical scenario in the absence of an infectious etiology. Paraneoplastic reactive arthritis is to be considered after excluding an underlying infection.

目的:说明一例系统性肥大细胞增多症在没有感染性病因的情况下模拟反应性关节炎。方法:查阅患者的医疗记录。结果:我们报告一个系统性肥大细胞增多症复发的病例,表现为全细胞减少并伴有膝关节单关节炎、膀胱炎和双侧结膜炎,在2-4天的同一时间间隔内同时发生,在没有感染性病因的情况下模拟反应性关节炎。结论:我们的病例表现出反应性关节炎的特征(尿道炎、结膜炎和关节炎三联征),没有传染性的触发,而是肥大细胞增多症的复发。在没有感染病因的情况下,面对这样的临床情况,我们应该跳出常规思维。副肿瘤反应性关节炎应在排除潜在感染后考虑。
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引用次数: 0
Use of Doxycycline in a Patient following Minocycline-Induced Lupus. 多西环素在二甲胺四环素诱发狼疮患者中的应用。
Pub Date : 2023-01-01 DOI: 10.1155/2023/7353644
Katherine Quinn Newman, Charles Guy Castles

Minocycline, a tetracycline antibiotic, is commonly used to treat rosacea and acne vulgaris. A rare adverse reaction of minocycline use is the development of drug-induced lupus. Fortunately, most patients recover from minocycline-induced lupus (MIL) after the drug is discontinued. However, many patients, after recovering from MIL, may desire further treatment for their acne and may consider doxycycline, a close relative of minocycline. Though no cases of doxycycline-induced lupus have been reported, there is little guidance in the medical literature as to whether doxycycline poses a particular risk to patients who have recovered from MIL. We report the long-term follow-up of a patient who recovered from MIL (the diagnosis satisfying clinical and laboratory criteria) and was treated for 8 years with various forms of doxycycline without any untoward effects, suggesting that, at least in some cases, doxycycline can be used safely following MIL.

二甲胺四环素是一种四环素抗生素,通常用于治疗酒渣鼻和寻常性痤疮。米诺环素使用的一个罕见的不良反应是药物性狼疮的发展。幸运的是,大多数患者在停药后从二甲胺四环素诱导的狼疮(MIL)中恢复。然而,许多患者,从MIL恢复后,可能希望进一步治疗他们的痤疮,并可能考虑多西环素,米诺环素的近亲。虽然没有多西环素诱发狼疮的病例报道,但在医学文献中很少有关于多西环素对MIL康复患者是否有特殊风险的指导。我们报告了一名MIL康复患者的长期随访(诊断符合临床和实验室标准),并使用各种形式的多西环素治疗8年,没有任何不良反应,这表明,至少在某些情况下,多西环素在MIL后可以安全使用。
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引用次数: 0
A 20-Year-Old Man with IgA Vasculitis following COVID-19 Vaccination. 1例20岁男性在接种COVID-19疫苗后出现IgA血管炎。
Pub Date : 2023-01-01 DOI: 10.1155/2023/9505383
Abdulaziz Alsubaie, Abdulmajeed Alshabanat, Abdulrahman Almizel, Mohammed Omair, Rahaf Alodaini

IgA vasculitis is a common type of vasculitis that is generally triggered by infectious causes. Vaccines have been reported as a trigger as well. Herein, we report a case of a young man who is previously healthy and who developed IgA vasculitis after the first dose of the COVID-19 mRNA vaccine Pfizer-BioNTech. The patient's symptoms were mainly skin and joint without renal or other system involvement. The patient had an excellent outcome with complete resolution after treatment with steroid tapering and azathioprine as a steroid-sparing agent over 6 months.

IgA血管炎是一种常见的血管炎,通常由感染性原因引起。据报道,疫苗也是一个触发因素。在此,我们报告了一例先前健康的年轻男性,在第一次接种COVID-19 mRNA疫苗辉瑞- biontech后出现IgA血管炎。患者的症状主要是皮肤和关节,没有肾脏或其他系统受累。在类固醇减量治疗和硫唑嘌呤作为类固醇保留剂治疗6个月后,患者有一个很好的结果,完全缓解。
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引用次数: 0
A Rare Case of Takayasu Arteritis Presenting as Pericarditis with Effusion. 罕见的高须动脉炎表现为心包炎并积液。
Pub Date : 2023-01-01 DOI: 10.1155/2023/6044765
Ian J Robertson, David K Mecham, Lisa M Conte, Michael F Loncharich

Takayasu arteritis (TAK) is a rare large-vessel vasculitis that is seen primarily in young females of Asian descent and is infrequently diagnosed in the United States. Pericardial effusion with or without pericarditis as a presenting feature of TAK is rare, with only about five percent of cases of pericarditis attributable to any autoimmune etiology. We present a case of a 22-year-old Caucasian woman who presented with a large, symptomatic pericardial effusion of unclear etiology, who after extensive laboratory workup and imaging to include whole-body positron emission tomography (PET) was diagnosed with TAK. In our patient, the use of whole-body PET showing characteristic hypermetabolism within the aortic arch helped secure our diagnosis while avoiding the need for pericardiocentesis. The patient had rapid symptomatic and radiographic improvement with the use of high-dose oral steroids in addition to colchicine and ibuprofen for her pericarditis and associated pericardial effusion. At follow-up just 1 week after initiation of steroids, only trace effusion was identified on transthoracic echocardiogram.

高须动脉炎(Takayasu arteritis, TAK)是一种罕见的大血管炎,主要见于年轻的亚裔女性,在美国很少被诊断出来。心包积液伴或不伴心包炎作为TAK的表现特征是罕见的,只有约5%的心包炎病例可归因于任何自身免疫性病因。我们报告一例22岁的白人女性,她表现出大量的症状性心包积液,病因不明,经过广泛的实验室检查和包括全身正电子发射断层扫描(PET)在内的影像学检查,被诊断为TAK。在我们的患者中,使用全身PET显示主动脉弓内特征性的高代谢有助于确保我们的诊断,同时避免了心包穿刺的需要。患者的心包炎和相关的心包积液在使用大剂量口服类固醇和秋水仙碱和布洛芬后,症状和影像学迅速改善。在类固醇治疗开始后1周的随访中,经胸超声心动图仅发现微量积液。
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引用次数: 0
A Case of an Elderly Woman Who Developed Corneal Perforation in the Clinical Course of Myeloperoxidase Positive Antineutrophil Cytoplasmic Antibody-Associated Vasculitis. 老年妇女髓过氧化物酶阳性抗中性粒细胞细胞质抗体相关血管炎临床过程中出现角膜穿孔1例。
Pub Date : 2023-01-01 DOI: 10.1155/2023/4246075
Shuhei Kobayashi, Makoto Harada, Aiko Yamada, Yasuhiro Iesato, Koji Hashimoto, Yuji Kamijo

Antineutrophil cytoplasmic antibody- (ANCA-) associated vasculitis (AAV) is a systemic vasculitis characterized by ANCA positivity and categorized into three main types: microscopic polyangiitis, granulomatosis with polyangiitis, and eosinophilic granulomatous with polyangiitis. Although AAV leads to systemic organ injury, such as of the lungs, kidneys, nerves, and skin, patients with AAV sometimes develop ocular lesions. Here, we report the case of an elderly woman who had been treated for AAV for seven years. She developed scleritis and relapsed twice, with elevation of serum disease markers such as ANCA titer and C-reactive protein. After the decline of these markers due to treatment with additional medication, her scleritis relapsed again and caused a corneal ulcer, which resulted in perforation without obvious marker elevation. She did not present with any symptoms of organ injury, except for ocular lesions. She was treated with surgery, followed by methylprednisolone and rituximab therapy. Subsequently, her ocular lesions and symptoms improved, and she did not relapse. AAV can cause various ocular manifestations. Although C-reactive protein and ANCA titers are useful markers of disease activity and the relapse of AAV complications, including ocular lesions, these markers do not always increase at the time of worsening ocular lesions. Therefore, it is important for clinicians treating patients with AAV to pay careful attention to serum data and physical findings, including the eyes.

抗中性粒细胞胞浆抗体- (ANCA-)相关性血管炎(AAV)是一种以ANCA阳性为特征的全体性血管炎,分为显微多血管炎、肉芽肿性多血管炎和嗜酸性肉芽肿性多血管炎三种主要类型。虽然AAV可导致全身器官损伤,如肺、肾、神经和皮肤,但AAV患者有时会出现眼部病变。在这里,我们报告的情况下,一位老年妇女谁已治疗AAV七年。患者出现巩膜炎并复发两次,血清疾病标志物如ANCA滴度和c反应蛋白升高。在这些标记物经过额外的药物治疗下降后,她的巩膜炎再次复发并引起角膜溃疡,导致穿孔,但标记物没有明显升高。除眼部病变外,无其他器官损伤症状。她接受手术治疗,随后甲基强的松龙和利妥昔单抗治疗。随后,她的眼部病变和症状改善,她没有复发。AAV可引起多种眼部表现。虽然c反应蛋白和ANCA滴度是疾病活动性和AAV并发症(包括眼部病变)复发的有用标记,但这些标记并不总是在眼部病变恶化时增加。因此,临床医生在治疗AAV患者时,必须仔细关注血清数据和包括眼睛在内的身体检查结果。
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引用次数: 0
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Case Reports in Rheumatology
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