a Department of Cardiology, Fuwai Hospital, National Center for Cardiovascular Diseases , Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing , China.
b McKusick-Zhang Center for Genetic Medicine, State Key Laboratory of Medical Molecular Biology, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing , China.
Clin Exp Hypertens. 2018;40(2):107-111. doi: 10.1080/10641963.2017.1334799. Epub 2017 Jul 18.
Liddle syndrome is an autosomal dominant form of monogenic hypertension. Phenotypic variability makes it difficult to identify patients with Liddle syndrome, resulting in misdiagnosis and severe complications at early age.
To identify mutation in SCNN1B and SCNN1G genes in an adolescent with suspicious Liddle syndrome and his family members and to explore the screening target subjects of Liddle syndrome.
Genetic analysis of the C-terminus of SCNN1B and SCNN1G genes was conducted in an adolescent, with treatment-resistant hypertension and hypokalemia, who was suspected of having Liddle syndrome, and his family members. A Medline research of the reported cases with Liddle syndrome was also performed.
A recurrent SCNN1B mutation, c.1853C>A (p.P618H), was detected in the 19-year-old male patient, and family screening identified five additional members who were heterozygous for the mutation. The diagnosis of Liddle syndrome was made in all affected individuals. Despite the phenotypic variability, a systematic review of 54 reported index cases revealed the early-onset hypertension, aged no more than 30 years, as a common feature.
Genetic screening for Liddle syndrome should be considered in hypertensive subjects with early penetrance, maybe no more than 30 years, after exclusion of common secondary causes of hypertension.
Liddle 综合征是一种常染色体显性遗传形式的单基因高血压。表型的可变性使得难以识别 Liddle 综合征患者,导致误诊和早期出现严重并发症。
鉴定一位疑似 Liddle 综合征的青少年及其家庭成员中 SCNN1B 和 SCNN1G 基因的突变,并探讨 Liddle 综合征的筛查目标人群。
对一位有治疗抵抗性高血压和低钾血症、疑似 Liddle 综合征的 19 岁男性青少年及其家庭成员的 SCNN1B 和 SCNN1G 基因 C 末端进行基因分析。还对报道的 Liddle 综合征病例进行了 Medline 研究。
在这位 19 岁的男性患者中检测到了 SCNN1B 基因的一个反复出现的突变,c.1853C>A(p.P618H),并通过家族筛查鉴定出另外 5 位杂合突变的成员。所有受影响的个体均被诊断为 Liddle 综合征。尽管表型存在可变性,但对 54 例已报道的指数病例的系统回顾显示,早发高血压(发病年龄不超过 30 岁)是一个常见特征。
在排除常见的继发性高血压原因后,如果高血压患者的发病年龄较早(可能不超过 30 岁),则应考虑进行 Liddle 综合征的遗传筛查。