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血红蛋白SC病患者的结节病和皮肌炎。病例报告及文献综述。

Sarcoidosis and dermatomyositis in a patient with hemoglobin SC. A case report and literature review.

作者信息

Brateanu A C, Caracioni A, Smith H R

机构信息

Department of Medicine, Huron Hospital, Cleveland, Ohio 44112, USA.

出版信息

Sarcoidosis Vasc Diffuse Lung Dis. 2000 Jun;17(2):190-3.

Abstract

Although sarcoidosis has occasionally been associated with hemoglobinopathies, its association with dermatomyositis is extremely rare. A 24 y/o African American male with hemoglobin SC disease developed proximal weakness. He had heliotrope rash, muscle weakness, elevated serum CK, myositis by EMG, and no malignancy. A muscle biopsy confirmed dermatomyositis. Two years later, he was hospitalized with constitutional and respiratory symptoms and parotid enlargement. Chest X-ray and CT scan showed diffuse micronodular infiltration in both lungs and mediastinal lymphadenopathy. A transbronchial lung biopsy revealed non-caseating granulomata consistent with sarcoidosis. AFB and fungal stains and cultures were negative. Thus, in this case, sarcoidosis developed after or in association with dermatomyositis. A review of world's literature showed five other non-identical cases of dermatomyositis associated with sarcoidosis.

摘要

虽然结节病偶尔与血红蛋白病相关,但它与皮肌炎的关联极为罕见。一名患有血红蛋白SC病的24岁非裔美国男性出现近端肌无力。他有向阳疹、肌肉无力、血清肌酸激酶升高、肌电图显示肌炎,且无恶性肿瘤。肌肉活检确诊为皮肌炎。两年后,他因全身症状、呼吸道症状和腮腺肿大住院。胸部X线和CT扫描显示双肺弥漫性微小结节浸润及纵隔淋巴结肿大。经支气管肺活检显示与结节病相符的非干酪样肉芽肿。抗酸杆菌和真菌染色及培养均为阴性。因此,在该病例中,结节病在皮肌炎之后发生或与之相关。对世界文献的回顾显示还有另外5例皮肌炎合并结节病的不同病例。

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