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Case reports in rheumatology 43건

  1. [해외논문]   Efficacy of Cyclosporine in the Induction and Maintenance of Remission in a Systemic Lupus Erythematosus Patient Presenting with Macrophage-Activating Syndrome  

    Cruz-Pé (Division of Rheumatology, Department of Medicine, University of Puerto Rico Medical Sciences Campus, San Juan, PR, USA ) , rez, Franchesca (Division of Rheumatology, Department of Medicine, University of Puerto Rico Medical Sciences Campus, San Juan, PR, USA ) , Vilá (Division of Rheumatology, Department of Medicine, University of Puerto Rico Medical Sciences Campus, San Juan, PR, USA ) , , Salvador (Division of Rheumatology, Department of Medicine, University of Puerto Rico Medical Sciences Campus, San Juan, PR, USA) , Rí , os, Grissel , Vilá , , Luis M.
    Case reports in rheumatology v.2018 ,pp. 1 - 4 , 2018 , 2090-6889 ,

    초록

    Macrophage-activating syndrome (MAS) is a rare condition characterized by dysfunctional macrophage activation leading to overproduction of cytokines and phagocytosis of erythrocytes, leukocytes, and platelets. MAS is associated with infectious diseases, malignancies, and autoimmune rheumatic disorders. Herein, we present a 22-year-old Hispanic woman with SLE who was hospitalized because of a three-week history of fever, fatigue, polyarthralgia, nausea, and abdominal pain. Initial laboratories showed severe pancytopenia with marked elevation of liver enzymes and ferritin levels. Bone marrow biopsy revealed macrophages with engulfed erythrocytes consistent with MAS. The patient was treated with high-dose corticosteroids, intravenous immunoglobulins, and cyclosporine 3 mg/kg/day. She had a remarkable clinical response to this therapy. She was continued on cyclosporine, and prednisone dose was gradually decreased to 7.5 mg daily without experiencing recurrent disease. She remained in full clinical remission for 12 months. Our case, together with other reports, suggests that combination therapy with corticosteroids, immunoglobulins, and cyclosporine appears to be effective for patients with SLE-associated MAS. Furthermore, cyclosporine seems to be a good drug for maintenance of remission.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  2. [해외논문]   A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment  

    Filippini, M. (Unità) , Cartella, S. (Operativa di Reumatologia, Spedali Civili di Brescia, Brescia, Italy ) , Bonzanini, O. (Unità) , Morello, E. (Operativa di Reumatologia, Spedali Civili di Brescia, Brescia, Italy ) , Tincani, A. (Unità)
    Case reports in rheumatology v.2018 ,pp. 1 - 3 , 2018 , 2090-6889 ,

    초록

    A 38-year-old woman was referred to our hospital for rheumatologic manifestations (migrant arthritis and tenosynovitis), without psoriasis or family history of psoriasis, gastroenteric manifestations, or recent genitourinary infections. The instrumental and laboratory tests have suggested a diagnosis of undifferentiated seronegative HLA-B27-positive spondyloarthritis with predominantly peripheral involvement. The symptoms were very severe and resistant to anti-inflammatory drugs and steroids. She had a history of hyaline-vascular unicentric Castleman's disease (HBV, HIV, and HHV-8 negative) treated with surgery resection. After a first pharmacological attempt with sulfasalazine (suspended for urticarial rash), we managed the patient with monotherapy tocilizumab 8 mg/kg, with full response of rheumatologic manifestations. The efficacy of tocilizumab was confirmed even after a follow-up of three years. Our experience seems to describe a new late-onset autoimmune disease (only 21 cases described in literature) potentially related to Castleman's disease. The patient experienced marked improvement from IL-6-based therapy (tocilizumab).

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  3. [해외논문]   Hemorrhagic Tamponade as Initial Manifestation of Systemic Lupus with Subsequent Refractory and Progressive Lupus Myocarditis Resulting in Cardiomyopathy and Mitral Regurgitation  

    Marijanovich, Nicole (Internal Medicine Department, Beaumont Hospital, Royal Oak, MI, USA ) , Halalau, Alexandra (Internal Medicine Department, Beaumont Hospital, Royal Oak, MI, USA)
    Case reports in rheumatology v.2018 ,pp. 1 - 4 , 2018 , 2090-6889 ,

    초록

    Systemic lupus erythematosus (SLE) is a heterogeneous autoimmune disease with a wide range of clinical and serological manifestations. Cardiac disease among patients with SLE is common and can involve the pericardium, myocardium, valves, conduction system, and coronary arteries. We are reporting a case of SLE in a young woman that is unique is unique in that initial symptoms consisted of pericarditis and hemorrhagic tamponade which remained progressive and resistant to aggressive immunosuppressive treatment and led to severe cardiomyopathy (ejection fraction of 25%) and severe (+4) mitral regurgitation. Her immunosuppressive treatment included hydroxychloroquine, high-dose steroids, intravenous immunoglobulins, azathioprine, and mycophenolate mofetil. Her disease progression was felt to be due to underlying uncontrolled SLE because the complement levels remained persistently low throughout the entire course and PET Myocardial Perfusion and Viability study showed stable persistent active inflammation. Eventually, she was treated with cyclophosphamide which led to improvement in ejection fraction to 55% with only mild mitral regurgitation.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  4. [해외논문]   ANCA Vasculitis and Hemophagocytic Lymphohistiocytosis following a Fecal Microbiota Transplant  

    Amlani, Adam (Department of Internal Medicine, University of Calgary, Calgary, AB, Canada ) , Bromley, Amy (Department of Pathology, University of Calgary, Calgary, AB, Canada ) , Fifi-Mah, Aurore (Department of Internal Medicine, Division of Rheumatology, University of Calgary, Calgary, AB, Canada)
    Case reports in rheumatology v.2018 ,pp. 1 - 3 , 2018 , 2090-6889 ,

    초록

    A 69-year-old female with antisynthetase syndrome, a history of multiple recurrent infections, and documented previous negative titres for anti-neutrophil cystoplasmic antibody (ANCA) suddenly developed a de novo MPO-ANCA-associated glomerulonephritis three weeks after a fecal microbiota transplantation (FMT) for recurrent Clostridium difficile infections. Six months following her FMT and less than two weeks following treatment for urosepsis, she developed severe cholestasis, a markedly elevated ferritin and hypertriglyceridemia. An initial liver biopsy was suggestive of drug-induced liver injury and thus she was treated with supportive care. After she failed to improve, a second liver biopsy supported the diagnosis of hemophagocytic lymphohistiocytosis (HLH). This case highlights difficulties surrounding the early diagnosis of HLH and also questions the role of FMT and/or recurrent infections as a trigger for ANCA-associated vasculitis.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  5. [해외논문]   Recurrent Migratory Transient Bone Marrow Edema of the Knees Associated with Low Vitamin D and Systemic Low Bone Mineral Density: A Case Report and Literature Review  

    Alsaed, Omar (Hamad Medical Corporation, Hamad General Hospital, Rheumatology Section, P.O. Box 3050, Doha, Qatar ) , Hammoudeh, Mohammad (Hamad Medical Corporation, Hamad General Hospital, Rheumatology Section, P.O. Box 3050, Doha, Qatar)
    Case reports in rheumatology v.2018 ,pp. 1 - 4 , 2018 , 2090-6889 ,

    초록

    Transient bone marrow edema (TBME) is a self-limiting disease characterized by joint pain with localized bone marrow edema by MRI and has been reported in many case series and case reports. It is well known that joints of the lower extremity including hips, knees, ankles, and feet are the classical sites for TBME. Many theories have been proposed for the pathogenesis of TBME. Systemic osteopenia and vitamin D deficiency is one of the theories that have been suggested in the last few years. In this case report, we present a middle-aged male patient, who presented with 4 attacks of TBME in both knees between September 2016 and August 2017. The patient was found to have persistently low vitamin D and osteopenic T score in DXA scan of the lumbar spine and hips. Patients of TBME usually present with joint pain that is provoked by weight-bearing physical activity. The aim of this case report is to raise the awareness that TBME can be the initial presentation of systemic loss of bone mineral density.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  6. [해외논문]   SjOgren Syndrome Complicated with Cystic Lung Disease and Pulmonary Amyloidosis  

    Takahashi, Koichiro (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan ) , Sadamatsu, Hironori (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan ) , Ogusu, Shinsuke (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan ) , Komiya, Kazutoshi (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan ) , Nakamura, Tomomi (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan ) , Kimura, Shinya (Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Sag) , Sueoka-Aragane, Naoko
    Case reports in rheumatology v.2018 ,pp. 1 - 5 , 2018 , 2090-6889 ,

    초록

    A 72-year-old Japanese woman was noted to have multiple cystic lung shadows and infiltrates on chest radiography and computed tomography (CT). She complained of dryness of the mouth and eyes, but she did not have respiratory symptoms, such as cough, sputum production, and dyspnea. Her laboratory findings showed high titers of anti-SSA/Ro and anti-SSB/La antibodies. Surgical lung biopsy was performed and demonstrated pathologic findings of amyloid light-chain deposition and bronchiolitis with lymphocytic infiltration. Taken altogether, she was diagnosed as SjOgren syndrome with bronchiolitis and pulmonary amyloidosis. Since then, she has been carefully followed up without treatment. After 6 years, the cystic lung lesions on CT gradually enlarged and increased in number, but she remained to have no respiratory symptoms and no manifestations of lymphoma. Here, we report a rare case of SjOgren syndrome complicated with cystic lung disease and pulmonary amyloidosis.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  7. [해외논문]   Erosive Arthritis, Fibromatosis, and Keloids: A Rare Dermatoarthropathy  

    Aslam, Fawad (Division of Rheumatology, Department of Internal Medicine, Mayo Clinic, Scottsdale, AZ, USA ) , Flug, Jonathan A. (Department of Radiology, Mayo Clinic, Scottsdale, AZ, USA ) , Yonan, Yousif (Department of Dermatology, Mayo Clinic, Scottsdale, AZ, USA ) , Noland, Shelley S. (Division of Plastic Surgery, Department of Surgery, Mayo Clinic Hospital, Phoenix, AZ, USA)
    Case reports in rheumatology v.2018 ,pp. 1 - 5 , 2018 , 2090-6889 ,

    초록

    Polyfibromatosis is a rare disease characterized by fibrosis manifesting in different locations. It is commonly characterized by palmar fibromatosis (Dupuytren's contracture) in variable combinations with plantar fibromatosis (Ledderhose's disease), penile fibromatosis (Peyronie's disease), knuckle pads, and keloids. There are only three reported cases of polyfibromatosis and keloids with erosive arthritis. We report one such case and review the existing literature on this rare syndrome.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  8. [해외논문]   Axial Spondyloarthritis and Autosomal Dominant Polycystic Kidney Disease in Two Siblings: A Rare Cooccurrence  

    Yurdakul, Ozan Volkan (Department of Physical Medicine and Rehabilitation, Bezmialem Foundation University, Istanbul, Turkey ) , Tamer, Abdulkerim Furkan (Department of Nephrology, Gaziosmanpasa Taksim Training and Research Hospital, Istanbul, Turkey ) , Kü (Department of Physical Medicine and Rehabilitation, Bezmialem Foundation University, Istanbul, Turkey ) , ç (Department of Physical Medicine and Rehabilitation, Bezmialem Foundation University, Istanbul, Turkey) , ü , kakkaş , , Okan , Rezvani, Aylin
    Case reports in rheumatology v.2018 ,pp. 1 - 6 , 2018 , 2090-6889 ,

    초록

    Autosomal dominant polycystic kidney disease (ADPKD) is the most frequently occurring hereditary kidney disease, and axial spondyloarthritis (SpA) is one of the most frequently occurring rheumatic diseases. Treatment-related decisions for axial SpA may pose a challenge in case of renal involvement. The authors describe two siblings with cooccurrence of these two diseases. The association of these two diseases is not well known. Practitioners should monitor renal function in SpA patients and take treatment-related decisions regarding renal involvement. Antitumor necrosis factor-alpha (anti-TNF- α ) agents may be used in case nonsteroidal anti-inflammatory drugs (NSAIDs) cannot be utilized.

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    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  9. [해외논문]   Cardiogenic Shock: An Unusual Initial Presentation of Churg-Strauss Syndrome  

    Apirami, M. (Teaching Hospital Jaffna, Jaffna, Sri Lanka ) , Pratheepan, J. A. (Teaching Hospital Jaffna, Faculty of Medicine, University of Jaffna, Jaffna, Sri Lanka ) , Kumanan, T. (Teaching Hospital Jaffna, Faculty of Medicine, University of Jaffna, Jaffna, Sri Lanka ) , Guruparan, M. (Teaching Hospital Jaffna, Jaffna, Sri Lanka ) , Selvaratnam, G. (Teaching Hospital Jaffna, Faculty of Medicine, University of Jaffna, Jaffna, Sri Lanka)
    Case reports in rheumatology v.2018 ,pp. 1 - 3 , 2018 , 2090-6889 ,

    초록

    Churg-Strauss syndrome (CSS) is a rare autoimmune condition, characterized by necrotizing extravascular eosinophil rich granulomatous inflammation of the tissues and disseminated small-medium sized vessel vasculitis in a patient with bronchial asthma and tissue eosinophilia. Though pulmonary involvement is the predominant feature of CSS, extra pulmonary involvement, in particular, cardiac involvement, denotes an adverse outcome. Here we report a 50-year-old female who presented with cardiogenic shock due to an acute coronary event as the initial manifestation of CSS. A subsequent coronary angiogram revealed normal epicardial coronaries. She was a patient with bronchial asthma and developed vasculitic rash, bilateral sensory motor polyneuropathy, and migratory peripheral lung field shadows in the background of peripheral eosinophilia during the course of the illness. She was diagnosed as having CSS based on ACR criteria and aggressively treated with immunosuppressants according to her Five-Factor Score and has shown prompt response to therapy. This case report adds to the literature another rare initial presentation of CSS to the existing array of its clinical manifestations.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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  10. [해외논문]   A Case of Familial Mediterranean Fever with Extensive Lymphadenopathy and Complex Heterozygous Genotype Presenting in the Fourth Decade  

    Al-Khafaji, Jawad (Department of Medicine, Virginia Commonwealth University Health System, Richmond, VA 23298, USA ) , Ganz-Lord, Fran (CareMount Medical, Chappaqua, NY 10514, USA ) , Konjeti, Venkata Rajesh (Department of Medicine, Virginia Commonwealth University Health System, Richmond, VA 23298, USA ) , Viny, Aaron D. (Human Oncology & Pathogenesis Program, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA)
    Case reports in rheumatology v.2018 ,pp. 1 - 5 , 2018 , 2090-6889 ,

    초록

    Familial Mediterranean fever (FMF) is an inherited disease caused by loss of function mutations in the MEFV gene encoding pyrin, a negative regulator of interleukin-1. The disease is characterized by recurrent fever and self-limited attacks of joint, chest, and abdominal pain but lymphadenopathy is an infrequent manifestation. While mesenteric lymphadenopathy has been described in several cases in the literature; hilar, paratracheal, axillary, pelvic, and retroperitoneal lymphadenopathy are extremely rare and have been reported separately in very few individuals. In this report, we present a patient with late-onset FMF with extensive lymphadenopathy in all of the aforementioned anatomic regions. Genetic analysis identified three heterozygous pyrin mutations in a patient with no affected family members. Genetic investigation of the patient's mother identified a novel carrier haplotype E148Q/P369S. The proband also inherited the previously described and rare A744S mutation previously not thought to be a disease-defining lesion. This unique compound heterozygous genotype resulted in a novel genotype-phenotype association producing an atypical clinical presentation of FMF that fits within the pattern of several case reports of late-onset disease with respect to clinical course and therapeutic response.

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    회원님의 원문열람 권한에 따라 열람이 불가능 할 수 있으며 권한이 없는 경우 해당 사이트의 정책에 따라 회원가입 및 유료구매가 필요할 수 있습니다.이동하는 사이트에서의 모든 정보이용은 NDSL과 무관합니다.

    NDSL에서는 해당 원문을 복사서비스하고 있습니다. 아래의 원문복사신청 또는 장바구니담기를 통하여 원문복사서비스 이용이 가능합니다.

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