Moreno-Carranza B, Gentsch M, Stein S, Schambach A, Santilli G, Rudolf E, Ryser M F, Haria S, Thrasher A J, Baum C, Brenner S, Grez M
Division of Applied Virology and Gene Therapy, Institute for Biomedical Research, Georg-Speyer-Haus, Frankfurt, Germany.
Gene Ther. 2009 Jan;16(1):111-8. doi: 10.1038/gt.2008.143. Epub 2008 Sep 11.
Gene therapy has proven to be of potential value for the correction of inherited hematopoietic disorders. However, the occurrence of severe side effects in some of the clinical trials has questioned the safety of this approach and has hampered the use of long terminal repeat-driven vectors for the treatment of a large number of patients. The development of self-inactivating (SIN) vectors with reduced genotoxicity provides an alternative to the currently used vectors. Our initial attempts to use SIN vectors for the correction of a myeloid disorder, chronic granulomatous disease, failed due to low vector titers and poor transgene expression. The optimization of the transgene cDNA (gp91(phox)) resulted in substantially increased titers and transgene expression. Most notably, transgene optimization significantly improved expression of a second cistron located downstream of gp91(phox). Thus, optimization of the transgene sequence results in higher expression levels and increased therapeutic index allowing the use of low vector copy numbers per transduced cell and weaker internal promoters.
基因治疗已被证明在纠正遗传性造血系统疾病方面具有潜在价值。然而,一些临床试验中出现的严重副作用引发了对这种治疗方法安全性的质疑,并阻碍了使用长末端重复序列驱动的载体治疗大量患者。具有降低基因毒性的自我失活(SIN)载体的开发为当前使用的载体提供了一种替代方案。我们最初尝试使用SIN载体纠正一种髓系疾病——慢性肉芽肿病,但由于载体滴度低和转基因表达不佳而失败。转基因cDNA(gp91(phox))的优化导致滴度和转基因表达大幅提高。最值得注意的是,转基因优化显著改善了位于gp91(phox)下游的第二个顺反子的表达。因此,转基因序列的优化导致更高的表达水平和更高的治疗指数,允许在每个转导细胞中使用低载体拷贝数和较弱的内部启动子。