A genetic defect resulting in mild low-renin hypertension.

نویسندگان

  • R C Wilson
  • S Dave-Sharma
  • J Q Wei
  • V R Obeyesekere
  • K Li
  • P Ferrari
  • Z S Krozowski
  • C H Shackleton
  • L Bradlow
  • T Wiens
  • M I New
چکیده

Severe low-renin hypertension has few known causes. Apparent mineralocorticoid excess (AME) is a genetic disorder that results in severe juvenile low-renin hypertension, hyporeninemia, hypoaldosteronemia, hypokalemic alkalosis, low birth weight, failure to thrive, poor growth, and in many cases nephrocalcinosis. In 1995, it was shown that mutations in the gene (HSD11B2) encoding the 11beta-hydroxysteroid dehydrogenase type 2 enzyme (11beta-HSD2) cause AME. Typical patients with AME have defective 11beta-HSD2 activity, as evidenced by an abnormal ratio of cortisol to cortisone metabolites and by an exceedingly diminished ability to convert [11-3H]cortisol to cortisone. Recently, we have studied an unusual patient with mild low-renin hypertension and a homozygous mutation in the HSD11B2 gene. The patient came from an inbred Mennonite family, and though the mutation identified her as a patient with AME, she did not demonstrate the typical features of AME. Biochemical analysis in this patient revealed a moderately elevated cortisol to cortisone metabolite ratio. The conversion of cortisol to cortisone was 58% compared with 0-6% in typical patients with AME whereas the normal conversion is 90-95%. Molecular analysis of the HSD11B2 gene of this patient showed a homozygous C-->T transition in the second nucleotide of codon 227, resulting in a substitution of proline with leucine (P227L). The parents and sibs were heterozygous for this mutation. In vitro expression studies showed an increase in the Km (300 nM) over normal (54 nM). Because approximately 40% of patients with essential hypertension demonstrate low renin, we suggest that such patients should undergo genetic analysis of the HSD11B2 gene.

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

APPARENT MINERALOCORTICOID EXCESS IN THREE SIBLINGS

Three siblings (1 boy, 2 girls) with hypertension and hypokalemia are presented, two with low plasma aldosterone and suppressed renin activity and the eldest with a high renin and aldosterone level due to secondary changes in her kidneys. Urinary tetrahydrocortisol (THF) was increased relative to tetrahydrocortisone (THE). Cortisol ring A reduction constant was also lower than normal. Thes...

متن کامل

A rare case of childhood hypertension with apparent mineralocorticoid excess syndrome

Hypertension is defined as average systolic blood pressure and/or diastolic blood pressure that are ≥95th percentile for gender, age, and height on ≥3 occasions (1). Renal parenchymal disease and essential hypertension are the most important causes in <12 and >12 years, respectively (2). Hypertension with hypokalaemia and suppression of plasma renin activity is known as mineralocorticoid hypert...

متن کامل

Increased central blood volume: a poosible pathophysiological factor im mild low-renin essential hypertension.

1. Patients with mild low-renin hypertension characteristically have increased central blood volume. The total blood volume is normal; the larger central blood volume reflects a shift of the blood from peripheral to central (cardiopulmonary) segments of the capacity system. 2. A relationship between central blood volume and plasma renin activity is demonstrable during tilting in normal and hype...

متن کامل

Liddle’s Syndrome

In 1963 Dr Grant Liddle, an endocrinologist in the United States described this syndrome. It is a rare inherited disorder of sodium channels resulting in excessive salt reabsorption from the distal nephron [1]. Initial presentation has been described from infancy through to late adulthood and in some circumstances first presentation of genetically prone patients has been evident with pregnancy ...

متن کامل

Familial aggregation of low-renin hypertension.

Low-renin hypertension, representing roughly one quarter of all essential hypertension, is widely recognized by distinct physiological features, including salt-sensitivity, diuretic- responsiveness, and a favorable natural history. Although certain demographic features including age, ethnicity, and diabetes mellitus predispose to low-renin hypertension, these factors account for only a minority...

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

عنوان ژورنال:
  • Proceedings of the National Academy of Sciences of the United States of America

دوره 95 17  شماره 

صفحات  -

تاریخ انتشار 1998