Bordignon C, Notarangelo L D, Nobili N, Ferrari G, Casorati G, Panina P, Mazzolari E, Maggioni D, Rossi C, Servida P, Ugazio A G, Mavilio F
Telethon Gene Therapy Program for Genetic Diseases, DIBIT, Istituto Scientifico H. S. Raffaele, Milan, Italy.
Science. 1995 Oct 20;270(5235):470-5. doi: 10.1126/science.270.5235.470.
Adenosine deaminase (ADA) deficiency results in severe combined immunodeficiency, the first genetic disorder treated by gene therapy. Two different retroviral vectors were used to transfer ex vivo the human ADA minigene into bone marrow cells and peripheral blood lymphocytes from two patients undergoing exogenous enzyme replacement therapy. After 2 years of treatment, long-term survival of T and B lymphocytes, marrow cells, and granulocytes expressing the transferred ADA gene was demonstrated and resulted in normalization of the immune repertoire and restoration of cellular and humoral immunity. After discontinuation of treatment, T lymphocytes, derived from transduced peripheral blood lymphocytes, were progressively replaced by marrow-derived T cells in both patients. These results indicate successful gene transfer into long-lasting progenitor cells, producing a functional multilineage progeny.
腺苷脱氨酶(ADA)缺乏会导致严重联合免疫缺陷,这是第一种通过基因疗法治疗的遗传性疾病。使用两种不同的逆转录病毒载体将人ADA小基因体外转移到两名接受外源性酶替代疗法的患者的骨髓细胞和外周血淋巴细胞中。经过2年的治疗,表达转移ADA基因的T淋巴细胞、B淋巴细胞、骨髓细胞和粒细胞实现了长期存活,免疫谱正常化,细胞免疫和体液免疫得以恢复。治疗中断后,两名患者中源自转导外周血淋巴细胞的T淋巴细胞逐渐被骨髓来源的T细胞取代。这些结果表明基因已成功转移到持久的祖细胞中,产生了具有功能的多谱系后代。