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依曲替酯治疗戈谢病的酶替代疗法:一项随机、双盲、多中心、3 期研究的结果。

Enzyme replacement therapy with velaglucerase alfa in Gaucher disease: Results from a randomized, double-blind, multinational, Phase 3 study.

机构信息

Sanatorio Español, Asunción, Paraguay.

出版信息

Am J Hematol. 2013 Mar;88(3):166-71. doi: 10.1002/ajh.23381. Epub 2013 Feb 6.

DOI:10.1002/ajh.23381
PMID:23386328
Abstract

Type 1 Gaucher disease (GD1), resulting from glucocerebrosidase deficiency, leads to splenomegaly, hepatomegaly, anemia, thrombocytopenia, and bone involvement. Current standard treatment is enzyme replacement therapy. Velaglucerase alfa is an enzyme replacement product for GD1, with the same amino acid sequence as naturally occurring human glucocerebrosidase. This multinational, Phase 3 trial evaluated the efficacy and safety of two doses of velaglucerase alfa in 25 treatment-naïve, anemic patients with GD1 (4-62 years of age), randomized to intravenous velaglucerase alfa 60 U/kg (n=12) or 45 U/kg body weight (n=13) every other week for 12 months. The primary endpoint was change from baseline in hemoglobin concentration in the 60 U/kg arm. At 12 months, mean hemoglobin concentrations increased from baseline [60 U/kg: +23.3%; +2.43 g/dL (P<0.001); 45 U/kg: +23.8%; +2.44 g/dL (P<0.001)], as did mean platelet counts [60 U/kg: +65.9%; +50.9 × 10(9) /L (P=0.002); 45 U/kg: +66.4%; +40.9 × 10(9) /L(P=0.01)]. Mean splenic volume decreased from baseline [60 U/kg: -50.4%, from 14.0 to 5.8 multiples of normal (MN) (P=0.003); 45 U/kg: -39.9%, from 14.5 to 9.5 MN (P=0.009)]. No drug-related serious adverse events or withdrawals were observed. One patient developed antibodies. Velaglucerase alfa was generally well tolerated and effective for adults and children with GD1 in this study. All disease-specific parameters measured demonstrated clinically meaningful improvements after 12 months.

摘要

类型 1 戈谢病(GD1)是由于葡萄糖脑苷脂酶缺乏引起的,导致脾肿大、肝肿大、贫血、血小板减少和骨骼受累。目前的标准治疗是酶替代疗法。维拉苷酶阿尔法是一种用于 GD1 的酶替代产品,其氨基酸序列与天然存在的人类葡萄糖脑苷脂酶相同。这项多中心、3 期试验评估了两种剂量的维拉苷酶阿尔法在 25 名未经治疗的贫血 GD1 患者(4-62 岁)中的疗效和安全性,这些患者随机接受静脉注射维拉苷酶阿尔法 60 U/kg(n=12)或 45 U/kg 体重(n=13),每两周一次,持续 12 个月。主要终点是 60 U/kg 组从基线血红蛋白浓度的变化。在 12 个月时,平均血红蛋白浓度从基线升高[60 U/kg:+23.3%;+2.43 g/dL(P<0.001);45 U/kg:+23.8%;+2.44 g/dL(P<0.001)],平均血小板计数也升高[60 U/kg:+65.9%;+50.9×10(9)/L(P=0.002);45 U/kg:+66.4%;+40.9×10(9)/L(P=0.01)]。平均脾脏体积从基线下降[60 U/kg:-50.4%,从 14.0 到 5.8 个正常倍数(MN)(P=0.003);45 U/kg:-39.9%,从 14.5 到 9.5 MN(P=0.009)]。未观察到与药物相关的严重不良事件或退出。一名患者产生了抗体。在这项研究中,维拉苷酶阿尔法对 GD1 成人和儿童总体耐受良好且有效。所有测量的疾病特异性参数在 12 个月后均显示出有临床意义的改善。

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