Ledley F D, Grenett H E, DiLella A G, Kwok S C, Woo S L
Science. 1985 Apr 5;228(4695):77-9. doi: 10.1126/science.3856322.
Phenylketonuria (PKU) is caused by a genetic deficiency of the enzyme phenylalanine hydroxylase (PAH). A full-length complementary DNA clone of human PAH was inserted into a eukaryotic expression vector and transferred into mouse NIH3T3 cells which do not normally express PAH. The transformed mouse cells expressed PAH messenger RNA, immunoreactive protein, and enzymatic activity that are characteristic of the normal human liver products, demonstrating that a single gene contains all of the necessary genetic information to code for functional PAH. These results support the use of the human PAH probe in prenatal diagnosis and detection of carriers, to provide new opportunities for the biochemical characterization of normal and mutant enzymes, and in the investigation of alternative genetic therapies for PKU.
苯丙酮尿症(PKU)是由苯丙氨酸羟化酶(PAH)的基因缺陷引起的。将人PAH的全长互补DNA克隆插入真核表达载体,并转入通常不表达PAH的小鼠NIH3T3细胞。转化后的小鼠细胞表达了具有正常人肝脏产物特征的PAH信使RNA、免疫反应性蛋白和酶活性,这表明单个基因包含编码功能性PAH所需的所有遗传信息。这些结果支持将人PAH探针用于产前诊断和携带者检测,为正常和突变酶的生化特性研究提供新机会,并用于研究苯丙酮尿症的替代基因疗法。