Miller J L, Donahue R E, Sellers S E, Samulski R J, Young N S, Nienhuis A W
National Heart, Lung and Blood Institute, Bethesda, MD 20892.
Proc Natl Acad Sci U S A. 1994 Oct 11;91(21):10183-7. doi: 10.1073/pnas.91.21.10183.
Effective gene therapy for the severe hemoglobin (Hb) disorders, sickle-cell anemia and thalassemia, will require an efficient method to transfer, integrate, and express a globin gene in primary erythroid cells. To evaluate recombinant adeno-associated virus (rAAV) for this purpose, we constructed a rAAV vector encoding a human gamma-globin gene (pJM24/vHS432A gamma). Its 4725-nucleotide genome consists of two 180-bp AAV inverted terminal repeats flanking the core elements of hypersensitive sites 2, 3, and 4 from the locus control region of the beta-globin gene cluster, linked to a mutationally marked A gamma-globin gene (A gamma) containing native promoter and RNA processing signals. CD34+ human hematopoietic cells were exposed to rAAV particles at a multiplicity of infection of 500-1000 and cultured in semisolid medium containing several cytokines. A reverse transcriptase polymerase chain reaction assay distinguished mRNA signals derived from transduced and endogenous human gamma-globin genes. Twenty to 40% of human erythroid burst-forming unit-derived colonies expressed the rAAV-transduced A gamma-globin gene at levels 4-71% that of the endogenous gamma-globin genes. The HbF content of pooled control colonies was 26%, whereas HbF was 40% of the total in pooled colonies derived from rAAV transduced progenitors. These data establish that rAAV containing elements from the locus control region linked to a gamma-globin gene are capable of transferring and expressing that gene in primary human hematopoietic cells resulting in a substantial increase in HbF content.
对于严重的血红蛋白(Hb)疾病,即镰状细胞贫血和地中海贫血,有效的基因治疗需要一种有效的方法,以便在原代红系细胞中转移、整合并表达珠蛋白基因。为了评估重组腺相关病毒(rAAV)用于此目的的效果,我们构建了一个编码人γ-珠蛋白基因的rAAV载体(pJM24/vHS432Aγ)。其4725个核苷酸的基因组由两个180 bp的AAV反向末端重复序列组成,这两个重复序列位于β-珠蛋白基因簇基因座控制区超敏位点2、3和4的核心元件两侧,并与一个含有天然启动子和RNA加工信号的经突变标记的Aγ-珠蛋白基因(Aγ)相连。将CD34+人造血细胞以500 - 1000的感染复数暴露于rAAV颗粒,并在含有多种细胞因子的半固体培养基中培养。逆转录聚合酶链反应测定法可区分源自转导的和内源性人γ-珠蛋白基因的mRNA信号。20%至40%源自人红系爆式形成单位的集落以相当于内源性γ-珠蛋白基因水平4% - 71%的水平表达rAAV转导的Aγ-珠蛋白基因。汇集的对照集落的HbF含量为26%,而源自rAAV转导祖细胞的汇集集落中HbF占总量的40%。这些数据表明,含有来自基因座控制区且与γ-珠蛋白基因相连元件的rAAV能够在原代人造血细胞中转移并表达该基因,从而使HbF含量大幅增加。