Department of Medicine, Division of Pulmonary and Critical Care Medicine, New York University School of Medicine, New York, NY, 10016, USA,
Appl Biochem Biotechnol. 2013 Oct;171(4):916-26. doi: 10.1007/s12010-013-0367-z. Epub 2013 Aug 2.
Genetic deficiency of acid alpha glucosidase (GAA) results in glycogen storage disease type II (GSDII) or Pompe's disease. To investigate whether we could generate a functional recombinant human GAA enzyme (tobrhGAA) in tobacco seeds for future enzyme replacement therapy, we subcloned the human GAA cDNA into the plant expression plasmid-pBI101 under the control of the soybean β-conglycinin seed-specific promoter and biochemically analyzed the tobrhGAA. Tobacco seeds contain the metabolic machinery that is more compatible with mammalian glycosylation-phosphorylation and processing. We found the tobrhGAA to be enzymatically active was readily taken up by GSDII fibroblasts and in white blood cells from whole blood to reverse the defect. The tobrhGAA corrected the enzyme defect in tissues at 7 days after a single dose following intraperitoneal (IP) administration in GAA knockout (GAA(-/-)) mice. Additionally, we could purify the tobrhGAA since it bound tightly to the matrix of Sephadex G100 and can be eluted by competition with maltose. These data demonstrate indirectly that the tobrhGAA is fully functional, predominantly proteolytically cleaved and contains the minimal phosphorylation and mannose-6-phosphate residues essential for biological activity.
酸性α-葡萄糖苷酶(GAA)的遗传缺陷导致糖原贮积症 II 型(GSDII)或庞贝病。为了研究我们是否可以在烟草种子中生成功能性重组人 GAA 酶(tobrhGAA)用于未来的酶替代治疗,我们将人 GAA cDNA 亚克隆到植物表达质粒-pBI101 中,受大豆β-伴球蛋白种子特异性启动子的控制,并对 tobrhGAA 进行了生化分析。烟草种子包含更适合哺乳动物糖基化-磷酸化和加工的代谢机制。我们发现 tobrhGAA 具有酶活性,很容易被 GSDII 成纤维细胞摄取,并在全血中的白细胞中逆转缺陷。在 GAA 敲除(GAA(-/-))小鼠中,单次腹腔内(IP)给药 7 天后,tobrhGAA 可纠正组织中的酶缺陷。此外,由于它与 Sephadex G100 的基质紧密结合,并且可以通过与麦芽糖竞争洗脱,因此可以纯化 tobrhGAA。这些数据间接表明 tobrhGAA 具有完全的功能,主要是蛋白水解切割,并包含生物活性所必需的最小磷酸化和甘露糖-6-磷酸残基。