Marcus S, Christensen E, Malm G
Environmental Medicine Unit, Karolinska Institutet, Huddinge, Sweden.
Hum Mutat. 1993;2(6):473-7. doi: 10.1002/humu.1380020608.
We have identified the mutations in the hypoxanthine phosphoribosyltransferase (hprt) gene in five patients with the Lesch Nyhan syndrome (LN) by direct sequencing of hprt cDNA and genomic DNA. Three of the mutations affect splicing of exons 1, 2, and 9, respectively, while two are missense mutations in exons 3 and 8. All 5 mutations result in profound hprt deficiency as measured in fibroblast lysates. However, small differences in the clinical phenotype are seen between the patients. All these mutations are unique and have not been reported previously.
我们通过对次黄嘌呤磷酸核糖基转移酶(hprt)cDNA和基因组DNA进行直接测序,确定了5例莱施-奈恩综合征(LN)患者的hprt基因突变。其中3个突变分别影响外显子1、2和9的剪接,另外2个是外显子3和8中的错义突变。在成纤维细胞裂解物中检测发现,所有5个突变均导致hprt严重缺乏。然而,这些患者的临床表型存在细微差异。所有这些突变都是独特的,此前未见报道。