Dighe Niraja, Khoury Maroun, Mattar Citra, Chong Mark, Choolani Mahesh, Chen Jianzhu, Antoniou Michael N, Chan Jerry K Y
Experimental Fetal Medicine Group, Department of Obstetrics and Gynecology, Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore.
Interdisciplinary Research Group in Infectious Diseases, Singapore-Massachusetts Institute of Technology Alliance for Research and Technology, Singapore, Singapore.
PLoS One. 2014 Aug 12;9(8):e104805. doi: 10.1371/journal.pone.0104805. eCollection 2014.
Hematopoietic Stem Cell (HSC) targeted gene transfer is an attractive treatment option for a number of hematopoietic disorders caused by single gene defects. However, extensive methylation of promoter sequences results in silencing of therapeutic gene expression. The choice of an appropriate promoter is therefore crucial for reproducible, stable and long-term transgene expression in clinical gene therapy. Recent studies suggest efficient and stable expression of transgenes from the ubiquitous chromatin opening element (UCOE) derived from the human HNRPA2B1-CBX3 locus can be achieved in murine HSC. Here, we compared the use of HNRPA2B1-CBX3 UCOE (A2UCOE)-mediated transgene regulation to two other frequently used promoters namely EF1α and PGK in human fetal liver-derived HSC (hflHSC). Efficient transduction of hflHSC with a lentiviral vector containing an HNRPA2B1-CBX3 UCOE-eGFP (A2UCOE-eGFP) cassette was achieved at higher levels than that obtained with umbilical cord blood derived HSC (3.1x; p<0.001). While hflHSC were readily transduced with all three test vectors (A2UCOE-eGFP, PGK-eGFP and EF1α-eGFP), only the A2-UCOE construct demonstrated sustained transgene expression in vitro over 24 days (p<0.001). In contrast, within 10 days in culture a rapid decline in transgene expression in both PGK-eGFP and EF1α-eGFP transduced hflHSC was seen. Subsequently, injection of transduced cells into immunodeficient mice (NOD/SCID/Il2rg-/-) demonstrated sustained eGFP expression for the A2UCOE-eGFP group up to 10 months post transplantation whereas PGK-eGFP and EF1α-eGFP transduced hflHSC showed a 5.1 and 22.2 fold reduction respectively over the same time period. We conclude that the A2UCOE allows a more efficient and stable expression in hflHSC to be achieved than either the PGK or EF1α promoters and at lower vector copy number per cell.
造血干细胞(HSC)靶向基因转移是治疗多种由单基因缺陷引起的造血系统疾病的一种有吸引力的治疗选择。然而,启动子序列的广泛甲基化会导致治疗性基因表达沉默。因此,选择合适的启动子对于临床基因治疗中可重复、稳定和长期的转基因表达至关重要。最近的研究表明,源自人类HNRPA2B1 - CBX3基因座的泛在染色质开放元件(UCOE)能在小鼠造血干细胞中实现转基因的高效稳定表达。在此,我们将HNRPA2B1 - CBX3 UCOE(A2UCOE)介导的转基因调控与另外两个常用启动子,即EF1α和PGK,在人胎肝来源的造血干细胞(hflHSC)中进行了比较。用含有HNRPA2B1 - CBX3 UCOE - eGFP(A2UCOE - eGFP)盒的慢病毒载体高效转导hflHSC,其转导水平高于脐血来源的造血干细胞(3.1倍;p<0.001)。虽然hflHSC能被所有三种测试载体(A2UCOE - eGFP、PGK - eGFP和EF1α - eGFP)轻易转导,但只有A2 - UCOE构建体在体外24天以上表现出持续的转基因表达(p<0.001)。相比之下,在培养10天内,PGK - eGFP和EF1α - eGFP转导的hflHSC中的转基因表达迅速下降。随后,将转导细胞注射到免疫缺陷小鼠(NOD/SCID/Il2rg - / - )体内,结果显示A2UCOE - eGFP组在移植后长达10个月内持续表达eGFP,而PGK - eGFP和EF1α - eGFP转导的hflHSC在同一时间段内分别下降了5.1倍和22.2倍。我们得出结论,与PGK或EF1α启动子相比,A2UCOE能在hflHSC中实现更高效稳定的表达,且每个细胞的载体拷贝数更低。