Kinney M C, Hummell D S, Villiger P M, Hourigan A, Rollins-Smith L, Glick A D, Lawton A R
Department of Pathology, Vanderbilt University Medical Center, Nashville, Tennessee 37232.
J Clin Immunol. 1994 Nov;14(6):382-90. doi: 10.1007/BF01546323.
A 21-month-old boy presented with a papular rash, lymphoadenopathy, and splenomegaly. He developed symmetric polyarthritis, fever, and progressive glomerulonephritis. Serologies for viral agents including HIV were negative. Antinuclear antibody was transiently positive, but no anti-DNA antibodies were present. CH50 and serum C3 values were low. Biopsies of skin, kidney, bone marrow, and lymph node were obtained. There was a perivascular and periadnexal lymphocytic infiltrate in the skin, with a normal epidermis. Renal biopsy showed proliferative mesangial glomerulonephritis. Bone marrow showed an increased number of plasma cells. Lymph node showed histologic changes described in multicentric Castleman's disease including marked follicular hyperplasia, vascular proliferation, and interfollicular expansion with numerous plasma cells. IL-6 mRNA was demonstrated in cells in the marginal zone and interfollicular regions of the node by in situ hybridization. Likewise, the serum IL-6 level was elevated during a clinical exacerbation of the patient's nephritis. These data suggest an underlying lymphoproliferative disorder, such as Castleman's disease, with overproduction of IL-6 resulting in systemic features of the disease, including glomerulonephritis.
一名21个月大的男孩出现丘疹性皮疹、淋巴结病和脾肿大。他发展为对称性多关节炎、发热和进行性肾小球肾炎。包括HIV在内的病毒病原体血清学检查均为阴性。抗核抗体短暂阳性,但不存在抗DNA抗体。CH50和血清C3值较低。获取了皮肤、肾脏、骨髓和淋巴结的活检样本。皮肤活检显示血管周围和附件周围有淋巴细胞浸润,表皮正常。肾活检显示系膜增生性肾小球肾炎。骨髓显示浆细胞数量增加。淋巴结显示出多中心Castleman病中描述的组织学变化,包括明显的滤泡增生、血管增生以及滤泡间扩张伴大量浆细胞。通过原位杂交在淋巴结边缘区和滤泡间区域的细胞中检测到IL-6 mRNA。同样,在患者肾炎临床加重期间血清IL-6水平升高。这些数据提示存在潜在的淋巴增殖性疾病,如Castleman病,IL-6过度产生导致了该疾病的全身特征,包括肾小球肾炎。