Li Huijun, Cao Peixuan, Zhu Xiangyu, Zhu Yujie, Wu Xing, Li Jie
Prenatal Diagnosis Center, the Affiliated Drum Tower Hospital of Nanjing University Medical School, Nanjing, Jiangsu 210008, China.
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2022 Sep 10;39(9):932-937. doi: 10.3760/cma.j.cn511374-20210715-00597.
To detect potential variants in eight Chinese pedigrees affected with autosomal dominant polycystic kidney disease (ADPKD) and provide prenatal diagnosis for two of them.
Whole exome sequencing and high-throughput sequencing were carried out to detect variants of PKD1 and PKD2 genes in the probands. Sanger sequencing was used to validate the variants, and their pathogenicity was predicted by searching the ADPKD and protein variation databases.
Eight PKD1 variants were detected, which have included five nonsense mutations and three missense mutations. Among these, four nonsense variants (PKD1: c.7555C>T, c.7288C>T, c.4957C>T, c.11423G>A) were known to be pathogenic, whilst one missense variant (PKD1: c.2180T>G) was classified as likely pathogenic. Three novel variants were detected, which included c.6781G>T (p.Glu2261*), c.109T>G (p.Cys37Gly) and c.8495A>G (p.Asn2832Ser). Prenatal testing showed that the fetus of one family has carried the same mutation as the proband, while the fetus of another family did not.
PKD1 variants, including three novel variants, have been identified in the eight pedigrees affected with ADPKD. Based on these results, prenatal diagnosis and genetic counseling have been provided.
检测8个常染色体显性多囊肾病(ADPKD)中国家系中的潜在变异,并为其中2个家系提供产前诊断。
对先证者进行全外显子测序和高通量测序以检测PKD1和PKD2基因的变异。采用Sanger测序验证变异,并通过检索ADPKD和蛋白质变异数据库预测其致病性。
检测到8个PKD1变异,其中包括5个无义突变和3个错义突变。其中,4个无义变异(PKD1:c.7555C>T、c.7288C>T、c.4957C>T、c.11423G>A)已知具有致病性,而1个错义变异(PKD1:c.2180T>G)被归类为可能致病。检测到3个新变异,分别为c.6781G>T(p.Glu2261*)、c.109T>G(p.Cys37Gly)和c.8495A>G(p.Asn2832Ser)。产前检测显示,一个家系的胎儿携带与先证者相同的突变,而另一个家系的胎儿未携带。
在8个ADPKD家系中鉴定出PKD1变异,包括3个新变异。基于这些结果,提供了产前诊断和遗传咨询。