Ohtani H, Imai H, Yasuda T, Wakui H, Komatsuda A, Hamai K, Miura A B
Third Department of Internal Medicine, Akita University School of Medicine, Japan.
Am J Kidney Dis. 1995 Sep;26(3):511-5. doi: 10.1016/0272-6386(95)90499-9.
A 59-year-old woman with retinal vein thrombosis and livedo racemosa had hematuria (4+) and proteinuria (1.7 g/day). Skin biopsy showed swollen blood vessel walls with infiltration of mononuclear cells, which were compatible with livedo racemosa (vasculitis). Magnetic resonance imaging (MRI) of the brain demonstrated multiple lacunar infarctions in the basal ganglia and white matter. Renal biopsy showed that small round cells had infiltrated into the interstitium, and a reticular structure was observed in the glomerular hilus. An amorphous substance composed of a single cell was present in the glomerular capillary lumen. Immunofluorescent study demonstrated the deposition of only IgA, in a segmental pattern differing from the diffuse global mesangial pattern seen in IgA nephropathy. After combined therapy including 40 mg/day prednisolone, 50 mg/day cyclophosphamide, antiplatelet drug, and anticoagulant was started, proteinuria and hematuria improved to 0.5 g/day and 2+, respectively, at the time of discharge. Sneddon's syndrome is a rare entity characterized by livedo racemosa and cerebrovascular lesions. In our patient with livedo racemosa, occlusion of cerebral blood vessels, and nephropathy with segmental immunoglobulin A (IgA) deposition, no antiphospholipid antibodies were detected on routine examination. Dermatologists, neurologists, psychiatrists, and nephrologists should be aware of the existence of Sneddon's syndrome with nephropathy (LI-O-N).
一名59岁患有视网膜静脉血栓形成和网状青斑的女性出现血尿(4+)和蛋白尿(1.7克/天)。皮肤活检显示血管壁肿胀,有单核细胞浸润,符合网状青斑(血管炎)表现。脑部磁共振成像(MRI)显示基底节和白质有多处腔隙性梗死。肾活检显示小圆细胞浸润到间质,肾小球 hilus 可见网状结构。肾小球毛细血管腔内存在由单个细胞组成的无定形物质。免疫荧光研究显示仅 IgA 呈节段性沉积,与 IgA 肾病中所见的弥漫性全肾小球系膜模式不同。在开始包括每天40毫克泼尼松龙、50毫克环磷酰胺、抗血小板药物和抗凝剂的联合治疗后,出院时蛋白尿和血尿分别改善至0.5克/天和2+。斯内登综合征是一种罕见的疾病,其特征为网状青斑和脑血管病变。在我们这位患有网状青斑、脑血管闭塞以及伴有节段性免疫球蛋白A(IgA)沉积的肾病患者中,常规检查未检测到抗磷脂抗体。皮肤科医生、神经科医生、精神科医生和肾科医生应意识到存在伴有肾病的斯内登综合征(LI - O - N)。