Toka Hakan R, Luft Friedrich C
Howard Hughes Medical Institute, Department of Genetics, Yale University School of Medicine, New Haven, CT, USA.
Semin Nephrol. 2002 Mar;22(2):81-8. doi: 10.1053/snep.2002.30206.
Monogenic or Mendelian forms of hypertension have ushered in a revolution in our knowledge. If we add information on syndromes involving low blood pressure, this knowledge base is doubled. Glucocorticoid-remediable aldosteronism, apparent mineralocorticoid excess, and mutations in the mineralocorticoid receptor gene have given us brilliant insights into mineralocorticoid-induced hypertension. The latter discovery has elucidated how mutations may modify the receptor sufficiently to allow erstwhile antagonists to have an agonistic action. The epithelial sodium channel (ENaC) has been elucidated. Gain-of-function mutations in the beta and gamma subunits of ENaC cause Liddle's syndrome. Loss-of-function mutations in all 3 subunits of ENaC cause hypotension (pseudohypoaldosteronism type I). Thus, all 3 subunits can be mutated, causing either hyper- or hypotension. Three loci have been described for Gordon's syndrome, pseudohypoaldosteronism type II; 2 members of the WNK (with no ly sine K) serine-threonine kinase family have recently been found to be responsible. Autosomal-dominant hypertension with brachydactyly features normal sodium and renin-angiotensin-aldosterone responses. The gene has been mapped to chromosome 12p. The condition is interesting because it may represent a novel neural form of hypertension. The elucidation of Mendelian blood pressure-regulatory disorders has been a resounding success.
单基因或孟德尔式高血压已引发了我们知识领域的一场革命。如果再加上有关涉及低血压综合征的信息,这个知识库就会翻倍。糖皮质激素可治性醛固酮增多症、表观盐皮质激素过多症以及盐皮质激素受体基因突变,让我们对盐皮质激素诱导的高血压有了深刻的认识。后一项发现阐明了突变如何充分改变受体,使以往的拮抗剂产生激动作用。上皮钠通道(ENaC)已被阐明。ENaC的β和γ亚基的功能获得性突变会导致利德尔综合征。ENaC所有3个亚基的功能丧失性突变会导致低血压(I型假性醛固酮增多症)。因此,所有3个亚基都可能发生突变,导致高血压或低血压。已描述了戈登综合征(II型假性醛固酮增多症)的3个基因座;最近发现WNK(无赖氨酸K)丝氨酸 - 苏氨酸激酶家族的2个成员与之有关。具有短指特征的常染色体显性高血压患者的钠及肾素 - 血管紧张素 - 醛固酮反应正常。该基因已被定位到12号染色体短臂。这种情况很有意思,因为它可能代表一种新型的神经性高血压。孟德尔式血压调节障碍的阐明取得了巨大成功。