Reiterová J, Stekrová J, Peters D J M, Kapras J, Kohoutová M, Merta M, Zidovská J
Department of Nephrology, General Hospital, Prague, Czech Republic.
Hum Mutat. 2002 May;19(5):573. doi: 10.1002/humu.9035.
Autosomal dominant polycystic kidney disease (ADPKD) is a genetically heterogeneous disease caused by mutations in at least three different loci. Mutations in the PKD2 gene are responsible for approximately 15% of the cases of the disease. We have screened 14 Czech families for mutation in the PKD2 gene. Clear evidence against linkage to the PKD1 gene was established by CA-repeat markers in five families. The disease could be linked to both genes according to linkage analysis in nine families but we have chosen these families because of the mild clinical course. An affected member from each family was analyzed by heteroduplex analysis (HA) and single strand conformation polymorphism (SSCP) for all 15 coding regions. Samples exhibiting shifted bands on HA or SSCP gels were sequenced. We detected five mutations (four new, and one which was previously described) and two polymorphisms. The four new mutations include one insertion, one deletion, one substitution (leading to premature translation stop), one amino acid substitution. Our results confirm that different point or small changes distributed throughout the PKD2 gene without clustering are responsible for the disease.
常染色体显性多囊肾病(ADPKD)是一种基因异质性疾病,由至少三个不同基因座的突变引起。PKD2基因突变约占该疾病病例的15%。我们对14个捷克家庭进行了PKD2基因突变筛查。通过CA重复标记在五个家庭中明确建立了与PKD1基因不连锁的证据。根据九个家庭的连锁分析,该疾病可能与两个基因都有关联,但我们选择这些家庭是因为其临床病程较轻。对每个家庭的一名患病成员的所有15个编码区进行了异源双链分析(HA)和单链构象多态性(SSCP)分析。对在HA或SSCP凝胶上显示条带迁移的样本进行测序。我们检测到五个突变(四个新突变和一个先前已描述的突变)以及两个多态性。这四个新突变包括一个插入、一个缺失、一个替代(导致过早翻译终止)、一个氨基酸替代。我们的结果证实,分布在PKD2基因中且无聚集现象的不同点突变或小变化是导致该疾病的原因。