BioMarin Pharmaceutical Inc., Novato, CA, USA.
BioMarin Pharmaceutical Inc., Novato, CA, USA.
Mol Ther. 2018 Feb 7;26(2):496-509. doi: 10.1016/j.ymthe.2017.12.009. Epub 2017 Dec 14.
Hemophilia A is an X-linked bleeding disorder caused by mutations in the gene encoding the factor VIII (FVIII) coagulation protein. Bleeding episodes in patients are reduced by prophylactic therapy or treated acutely using recombinant or plasma-derived FVIII. We have made an adeno-associated virus 5 vector containing a B domain-deleted (BDD) FVIII gene (BMN 270) with a liver-specific promoter. BMN 270 injected into hemophilic mice resulted in a dose-dependent expression of BDD FVIII protein and a corresponding correction of bleeding time and blood loss. At the highest dose tested, complete correction was achieved. Similar corrections in bleeding were observed at approximately the same plasma levels of FVIII protein produced either endogenously by BMN 270 or following exogenous administration of recombinant BDD FVIII. No evidence of liver dysfunction or hepatocyte endoplasmic reticulum stress was observed. Comparable doses in primates produced similar levels of circulating FVIII. These preclinical data support evaluation of BMN 270 in hemophilia A patients.
A 型血友病是一种 X 连锁出血性疾病,由编码凝血因子 VIII(FVIII)蛋白的基因突变引起。预防性治疗可减少患者的出血发作,或使用重组或血浆衍生的 FVIII 进行急性治疗。我们构建了一种含有 B 域缺失(BDD)FVIII 基因(BMN 270)的腺相关病毒 5 载体,该载体带有肝特异性启动子。将 BMN 270 注射到血友病小鼠中,可导致 BDD FVIII 蛋白的剂量依赖性表达,并相应地纠正出血时间和失血。在测试的最高剂量下,可实现完全纠正。在大约相同的 FVIII 蛋白血浆水平下,无论是通过 BMN 270 内源性产生还是通过重组 BDD FVIII 外源性给予,都观察到了类似的出血纠正。未观察到肝功能障碍或肝细胞内质网应激的证据。在灵长类动物中,类似剂量可产生类似水平的循环 FVIII。这些临床前数据支持在 A 型血友病患者中评估 BMN 270。