Williamson D J, Hooper M L, Melton D W
Department of Pathology, University Medical School, Edinburgh, UK.
J Inherit Metab Dis. 1992;15(4):665-73. doi: 10.1007/BF01799622.
Lesch--Nyhan syndrome is an X-linked disease caused by the deficiency of hypoxanthine phosphoribosyltransferase, an enzyme involved in the purine salvage pathways. It is characterized by severe gout, choreoathetosis, self-mutilatory behaviour and mental retardation. The derivation of mice genetically deficient in this enzyme may help to elucidate the pathogenesis of the neurological abnormality where previously models using drug administration to mimic the disorder have had to suffice.
莱施-奈恩综合征是一种X连锁疾病,由次黄嘌呤磷酸核糖转移酶缺乏引起,该酶参与嘌呤补救途径。其特征为严重痛风、舞蹈手足徐动症、自残行为和智力迟钝。对这种酶基因缺陷的小鼠进行培育,可能有助于阐明神经异常的发病机制,在此之前,使用药物给药来模拟该疾病的模型只能满足于此。