Schröder W, Wulff K, Wehnert M, Seidlitz G, Herrmann F H
Institute for Medical Genetics, Ernst-Moritz-Arndt-University, Greifswald, Germany.
Hum Mutat. 1994;4(2):128-31. doi: 10.1002/humu.1380040206.
Genomic DNA and cDNA from fibroblasts from nine unrelated German patients with X-linked iduronate-2-sulfatase (IDS) deficiency showing variable clinical manifestation were screened for point mutations and small structural aberrations. Direct sequencing revealed a splice mutation skipping exon A, one nonsense mutation, and five missense mutations concerning the exons B, F and I of the IDS gene. Several novel missense mutations were found: A68E, S426X, I485R, Q293H, and D478G. One of the point mutations eliminating a recognition site for the restriction enzyme MspI was used as a direct marker for a prenatal diagnosis. A relationship between type of mutation and clinical picture could not be recognized.
对9名患有X连锁艾杜糖醛酸-2-硫酸酯酶(IDS)缺乏症且临床表现各异的不相关德国患者的成纤维细胞中的基因组DNA和cDNA进行了点突变和小结构畸变筛查。直接测序揭示了一个跳过外显子A的剪接突变、一个无义突变以及5个与IDS基因外显子B、F和I相关的错义突变。发现了几个新的错义突变:A68E、S426X、I485R、Q293H和D478G。消除限制性内切酶MspI识别位点的一个点突变被用作产前诊断的直接标记。未发现突变类型与临床表现之间的关联。