家族性噬血细胞性淋巴组织细胞增生症中的自然细胞毒性损伤。
Natural cytotoxicity impairment in familial haemophagocytic lymphohistiocytosis.
作者信息
Aricò M, Nespoli L, Maccario R, Montagna D, Bonetti F, Caselli D, Burgio G R
机构信息
Department of Paediatrics, University of Pavia, Italy.
出版信息
Arch Dis Child. 1988 Mar;63(3):292-6. doi: 10.1136/adc.63.3.292.
Abstract
Ten children with the characteristic clinical and haematological features of haemophagocytic lymphohistiocytosis are reported. Four patients treated with a combination of drugs comprising etoposide, methotrexate, and steroids were in complete remission after 10 to 30 months. Natural cytotoxic mechanisms including natural killer cell activity, antibody dependent cell mediated cytotoxicity, lymphokine activated killer cell activity, and natural killer cell like activity were persistently absent or severely impaired in these four patients despite their clinical remission. Their parents and one healthy sibling also had impaired natural cytotoxic mechanisms. Constitutional impairment of natural cytotoxic mechanisms could be important in the pathogenesis of haemophagocytic lymphohistiocytosis.
摘要
报告了10例具有噬血细胞性淋巴组织细胞增生症特征性临床和血液学表现的儿童。4例接受依托泊苷、甲氨蝶呤和类固醇联合药物治疗的患者在10至30个月后完全缓解。尽管这4例患者临床缓解，但包括自然杀伤细胞活性、抗体依赖性细胞介导的细胞毒性、淋巴因子激活的杀伤细胞活性和自然杀伤细胞样活性在内的自然细胞毒性机制持续缺失或严重受损。他们的父母和一名健康的兄弟姐妹也有自然细胞毒性机制受损的情况。自然细胞毒性机制的先天性损害可能在噬血细胞性淋巴组织细胞增生症的发病机制中起重要作用。
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