Hypocomplementemic urticarial vasculitis with Jaccoud's arthropathy and valvular heart disease: case report and review of the literature
Hypocomplementemic urticarial vasculitis with Jaccoud's arthropathy and valvular heart disease: case report and review of the literature
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DOI：
10.1177/0961203308090113
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发表时间：
2008-01-01
期刊：
影响因子：
2.6
通讯作者：
Takasaki, Y.
中科院分区：
文献类型：
--
作者：
Amano, H.;Furuhata, N.;Takasaki, Y.
我们描述一位日本女性患者，并伴有低补体麻风血管炎、贾库德关节病和心脏瓣膜病。1996年，她患上关节炎，近侧指间关节肿胀，双臂麻风样血管炎，用糖皮质激素(强的松龙30毫克/天)治疗后痊愈。抗中性粒细胞胞浆抗体、抗核抗体和类风湿因子检测均为阴性。皮肤活检检查结果与‘白细胞破碎性血管炎’相一致。在10年的观察中，患者表现为多发性关节炎，导致手足关节进行性畸形(无软骨丢失或骨质侵蚀)，持续性荨麻疹，寒冷加重，并伴有低补体血症和进行性心脏瓣膜病伴二尖瓣返流。之前只有三份报告记录了五名患者的这种罕见的表现组合。
We describe a female Japanese patient with concomitant hypocomplementemic urticarial vasculitis, Jaccoud's arthropathy and valvular heart disease. In 1996, she developed arthritis with swelling of both proximal interphalangeal joints and Urticarial vasculitis on both arms that was resolved by administration of glucocorticoid (prednisolone 30 mg/day). Tests for antineutrophil cytoplasmic antibodies, antinuclear antibody and rheumatoid factor gave negative results. The findings of a skin biopsy examination were consistent with 'leukocytoclastic vasculitis'. During 10 years of observation, the patient manifested polyarthritis leading to progressive deformity of the joints of the hands and feet (without loss of cartilage or erosion of bone), persistent urticaria exacerbated by cold and accompanied by hypocomplementemia and progressive cardiac valvular disease with mitral valve regurgitation. There are only three reports described previously documenting five patients with this rare combination of manifestations.