Cosma Maria Pia, Pepe Stefano, Annunziata Ida, Newbold Robert F, Grompe Markus, Parenti Giancarlo, Ballabio Andrea
Telethon Institute of Genetics and Medicine (TIGEM), Naples, Italy.
Cell. 2003 May 16;113(4):445-56. doi: 10.1016/s0092-8674(03)00348-9.
In multiple sulfatase deficiency (MSD), a human inherited disorder, the activities of all sulfatases are impaired due to a defect in posttranslational modification. Here we report the identification, by functional complementation using microcell-mediated chromosome transfer, of a gene that is mutated in MSD and is able to rescue the enzymatic deficiency in patients' cell lines. Functional conservation of this gene was observed among distantly related species, suggesting a critical biological role. Coexpression of SUMF1 with sulfatases results in a strikingly synergistic increase of enzymatic activity, indicating that SUMF1 is both an essential and a limiting factor for sulfatases. These data have profound implications on the feasibility of enzyme replacement therapy for eight distinct inborn errors of metabolism.
在多种硫酸酯酶缺乏症(MSD)这一人类遗传性疾病中,由于翻译后修饰缺陷,所有硫酸酯酶的活性均受损。在此,我们报告通过微细胞介导的染色体转移进行功能互补鉴定出一个在MSD中发生突变且能够挽救患者细胞系中酶缺乏的基因。在远缘物种中观察到该基因的功能保守性,提示其具有关键的生物学作用。SUMF1与硫酸酯酶共表达导致酶活性显著协同增加,表明SUMF1既是硫酸酯酶的必需因子也是限制因子。这些数据对八种不同先天性代谢缺陷的酶替代疗法的可行性具有深远影响。