Lajoie G, Kumar S, Min K W, Silva F G
Department of Pathology, University of Oklahoma Health Sciences Center, Oklahoma City, USA.
Am J Surg Pathol. 1995 Sep;19(9):1021-8. doi: 10.1097/00000478-199509000-00005.
Castleman's disease (also called giant lymph node hyperplasia or angiofollicular lymph node hyperplasia) is a clinicopathological entity of unknown etiology. Two histologic patterns of lymph nodes are classically recognized: the hyaline-vascular and plasma-cell variants. Recently, multicentric Castleman's disease has emerged as a separate clinical entity manifested primarily by generalized lymphadenopathy and systemic manifestations, such as thrombocytopenia, hemolytic anemia, hepatosplenomegaly, altered liver function tests, central nervous system alterations, and autoimmune manifestations. A number of renal alterations have been described in association with the two pathological variants of Castleman's disease, but thrombotic microangiopathy has been previously reported only once in a patient with Castleman's disease. No renal biopsy was performed in that patient, although there was evidence of renal dysfunction. We report two cases of biopsy-proven renal thrombotic microangiopathy associated with multicentric Castleman's disease. In addition to having lymph node pathology characteristic of Castleman's disease, both patients presented with generalized lymphadenopathy and systemic manifestations, including acute renal failure, hypergammaglobulinemia, anemia, thrombocytopenia, and hypoalbuminemia. Autoantibodies were present in both patients, including antiphospholipid antibodies in one patient. The renal biopsies, examined by light, immunofluorescence, and electron microscopy, were diagnostic for renal thrombotic microangiopathy. The simultaneous development of two rather uncommon syndromes, multicentric Castleman's disease and renal thrombotic microangiopathy, suggests a possible link between Castleman's disease and renal thrombotic microangiopathy. Furthermore, we propose that the production of autoantibodies, in particular antiphospholipid antibodies, may lead to the development of thrombotic microangiopathy in some patients with multicentric Castleman's disease.
卡斯特曼病(也称为巨大淋巴结增生症或血管滤泡性淋巴结增生症)是一种病因不明的临床病理实体。经典地认识到淋巴结有两种组织学模式:透明血管型和浆细胞型。最近,多中心性卡斯特曼病已成为一种独立的临床实体,主要表现为全身性淋巴结肿大和全身症状,如血小板减少、溶血性贫血、肝脾肿大、肝功能检查异常、中枢神经系统改变和自身免疫表现。已经描述了许多与卡斯特曼病的两种病理类型相关的肾脏改变,但血栓性微血管病此前仅在一名卡斯特曼病患者中报道过一次。该患者未进行肾活检,尽管有肾功能不全的证据。我们报告两例经活检证实的与多中心性卡斯特曼病相关的肾血栓性微血管病。除了具有卡斯特曼病特征性的淋巴结病理外,两名患者均出现全身性淋巴结肿大和全身症状,包括急性肾衰竭、高球蛋白血症、贫血、血小板减少和低白蛋白血症。两名患者均存在自身抗体,其中一名患者存在抗磷脂抗体。通过光镜、免疫荧光和电子显微镜检查的肾活检对肾血栓性微血管病具有诊断意义。多中心性卡斯特曼病和肾血栓性微血管病这两种相当罕见综合征的同时出现,提示卡斯特曼病与肾血栓性微血管病之间可能存在联系。此外,我们提出自身抗体,特别是抗磷脂抗体的产生,可能导致一些多中心性卡斯特曼病患者发生血栓性微血管病。