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낮은 류마티스관절염 질병활성도를 동반한 불응성 Felty 증후군에서 Rituximab의 치료
장성혜 ( Sung Hae Chang ),양지애 ( Ji Ae Yang ),이상진 ( Sang Jin Lee ),박준원 ( Jun Won Park ),신기철 ( Ki Chul Shin ),이은봉 ( Eun Bong Lee ),송영욱 ( Yeong Wook Song ),이은영 ( Eun Young Lee ) 대한류마티스학회 2014 대한류마티스학회지 Vol.21 No.1
Felty syndrome (FS) is a rare manifestation in rheumatoid arthritis (RA) characterized by neutropenia and splenomegaly. Treatment for FS is not well established because there has been no randomized controlled study. A few recent reports found rituximab effective in patients with refractory FS. According to those reports, most patients with RA and FS had active arthritis. Here we report a case of a patient with glucocorticoid dependent and disease-modifying anti-rheumatic drugs (DMARDs) refractory FS and quiescent RA who was successfully treated with rituximab.
베체트병 환자에서 발생한 Lenticulostriate 동맥류 파열 1예
황성준 ( Seongjun Hwang ),장성혜 ( Sung Hae Chang ),정상완 ( Sang Wan Chung ),하유정 ( You Jung Ha ),강은하 ( Eun Ha Kang ),송영욱 ( Yeong Wook Song ),이윤종 ( Yun Jong Lee ) 대한류마티스학회 2015 대한류마티스학회지 Vol.22 No.5
베체트병의 환자에서 뇌동맥류는 흔치 않은 중추신경계침범 임상상이며, 특히 lenticulostriate 동맥류가 발생하는 경우는 매우 드물다. 저자들은 활동성 베체트병에의한 lenticulostriate 동맥류 파열로 발생한 급성 좌측 편마비환자에서 고용량 글루코코르티코이드제제 투여로 호전된 국내첫 번째 증례를 경험하였기에 문헌고찰과 함께 보고한다. Behcet`s disease (BD) is characterized by recurrent oro-genital ulcers, skin lesions, and intraocular inflammation, but can also affect various internal organs. Vascular BD usually presents with luminal stenosis, thrombosis, or aneurysm formation in aorta and peripheral arteries. However, intracranial artery involvement has been uncommonly reported in patients with BD and BD cases with lenticulostriate artery aneurysm have been rarely described in the English-language literature. We hereby reported the first case of a Korean BD patient presenting with a ruptured lenticulostriate artery aneurysm, who received medical treatment, and reviewed the literature on reported cases of BD with intracranial aneurysms. (J Rheum Dis 2015;22:317-321)
불응성 염증성 근병증 환자에 대한 Rituximab 치료
양지애 ( Ji Ae Yang ),이상진 ( Sang Jin Lee ),박준원 ( Jun Won Park ),권현미 ( Hyun Mi Kwon ),문진영 ( Jin Young Moon ),고동진 ( Dong Jin Ko ),장성혜 ( Sung Hae Chang ),박진균 ( Jin Kyun Park ),이은봉 ( Eun Bong Lee ),송영욱 ( Yeo 대한류마티스학회 2013 대한류마티스학회지 Vol.20 No.5
Objective. To assess the efficacy and safety of rituximab (RTX) on disease activity and muscle strength in patients with inflammatory myopathies refractory to conventional therapy. Methods. Four inflammatory myopathy patients who had been refractory to glucocorticoids, one or more immunosuppressive therapies and intravenous immunoglobulin were treated on an open-label basis. Each patient received two 500 mg doses of RTX 2 weeks apart in one cycle. In one patient who did not respond after the first cycle of RTX, the infusion schedule was modified by the physician. We measured muscle enzyme including CPK, LDH and assessed muscle strength individually to evaluate RTX response. Additionally anti-CD19 antibody was measured. Results. Three patients responded to the first cycle of RTX treatment with improvements in muscle enzyme and muscle strength, and then maintained physical function over the duration of several infusion cycles. In one patient, muscle enzyme did not decrease after the first cycle of RTX, and a high dose glucocorticoid was given. After modifying the treatment schedule with monthly RTX infusion, his muscle enzyme level and muscle strength improved. Anti-CD19 antibody decreased after RTX generally, but responses were variable. Herpes zoster infection occurred in two patients. Conclusion. Rituximab may be a therapeutic choice in refractory inflammatory myopathy. However a further trial is needed to confirm the efficacy and prove the safety.
Churg-Strauss 증후군에서 동반된 심근내막섬유증
김혜원 ( Hye Won Kim ),송영욱 ( Yeong Wook Song ),장성혜 ( Sung Hae Chang ),류한희 ( Han Hee Ryu ),송란 ( Ran Song ),이은봉 ( Eun Bong Lee ),손대원 ( Dae Won Son ),김유영 ( You Young Kim ) 대한류마티스학회 2010 대한류마티스학회지 Vol.17 No.3
Cardiac involvement is an important cause of mortality in patients with Churg-Strauss syndrome. The typical cardiac presentation of Churg-Strauss syndrome includes pericarditis, myocarditis, and cardiomyopathy. Endomyocardial fibrosis has rarely been described in patients with Churg-Strauss syndrome. We experienced a patient with Churg-Strauss syndrome who exhibited exertional dyspnea and endomyocardial fibrosis visualized as delayed enhancement on cardiac magnetic resonance imaging (MRI). After glucocorticoid treatment, the patient`s symptom resolved, and the eosinophil count decreased to normal. Nine months later, the delayed-enhanced lesion on the cardiac MRI nearly disappeared. Here, we report a case of endomyocardial fibrosis in a patient with Churg-Strauss syndrome with a literature review.
증례 : 류마티스 ; 강직성 척추염에 동반된 아밀로이드증이 TNF 억제제로 호전된 1예
이재현 ( Jae Hyun Lee ),문진영 ( Jinyoung Moon ),이현우 ( Hyun Woo Lee ),장성혜 ( Sung Hae Chang ),이은봉 ( Eun Bong Lee ),송영욱 ( Yeong Wook Song ),이은영 ( Eun Young Lee ) 대한내과학회 2015 대한내과학회지 Vol.89 No.2
Secondary amyloidosis (Amyloid A amyloidosis) occurs when serum amyloid S accumulates in various tissues and organs. It often accompanies chronic inflammatory disease, especially rheumatoid arthritis, as well as ankylosing spondylitis (AS) and Crohn`s disease. Although amyloid deposits were reported in 7% of the abdominal fatty tissue samples from AS patients, most of the patients had no symptoms. In a large study, 1.1% of 730 patients with AS were diagnosed with amyloidosis. Most of them showed renal involvement, while none had gastrointestinal symptoms. In South Korea, only a few cases of gastrointestinal involvement of secondary amyloidosis have been reported in AS patients, and there is no report on the effect of a tumor necrosis factor (TNF)-α inhibitor. We report a case of secondary amyloidosis in an AS patient who improved with the TNF-α inhibitor etanercept. (Korean J Med 2015;89:259-263)
이은봉 ( Eun Bong Lee ),김혜원 ( Hye Won Kim ),신기철 ( Ki Chul Shin ),송영욱 ( Yeong Wook Song ),임재준 ( Jae Joon Yim ),장성혜 ( Sung Hae Chang ),류한희 ( Han Hee Ryu ) 대한류마티스학회 2011 대한류마티스학회지 Vol.18 No.1
Caplan originally described distinctive pulmonary nodules in miners who had suffered from rheumatoid arthritis. Later, the pulmonary nodules, together with a history of rheumatoid arthritis and exposure to inorganic dust were called Caplan`s syndrome. This syndrome has been described by case reports in many countries but only two cases have been reported in Korea up to now. The patient in this case report was a 70-years-old man who had worked in the construction field for 20 years mainly demolishing buildings. He was diagnosed with silicosis by a lung biopsy 1 year prior to admission. He suddenly developed arthralgia and morning stiffness in multiple joints 2 weeks prior to admission. Chest imaging revealed aggravation of the bilateral pulmonary nodules. He was diagnosed with seropositive rheumatoid arthritis. The lung nodules, arthralgia, and morning stiffness improved clinically after treatment with the corticosteroid and disease modifying anti-rheumatic drugs.
대동맥주위염과 비후성 경뇌막염으로 발현한 베게너 육아종증의
이은봉 ( Eun Bong Lee ),이은영 ( Eun Young Lee ),이윤종 ( Yun Jong Lee ),김혜원 ( Hye Won Kim ),윤찬영 ( Chan Young Yun ),송영욱 ( Yeong Wook Song ),강은하 ( Eun Ha Kang ),장성혜 ( Sung Hae Chang ) 대한류마티스학회 2010 대한류마티스학회지 Vol.17 No.4
Wegener`s grandulomatosis (WG) is a systemic small-vessel vasculitis characterized by necrotizing granulomatous lesions and anti-neutrophil cytoplasmic antibodies (ANCA). WG frequently affects the upper and lower respiratory tracts and kidney but involvement of the pachymeninges or large vessels has rarely been reported. We report the first case of WG with periaortitis and pachymeningitis in Korea. A 57-year-old female complained of a stabbing headache, paresthesia below the thoracic 4th nerve level, and lower extremity weakness. During her course, she developed otitis media with sensorineural hearing loss, nodular scleritis, and microhematuria. A neuroimaging study showed an intradural mass in the area from the cervical 7th spine to the thoracic 5th spine, left cerebral convexity, and the left petrous area. A chest computed tomography scan revealed an enhanced and thickened aortic arch. A dural biopsy showed necrotizing granulomatous inflammation, and anti-MPO ANCA was positive. Under a diagnosis of WG, she received high-dose glucocorticoid and oral cyclophosphamide with improvement.