Literature DB >> 9643296

The diagnosis of Liddle syndrome by identification of a mutation in the beta subunit of the epithelial sodium channel.

S N Jackson1, B Williams, P Houtman, R C Trembath.   

Abstract

Hypertension is a common multifactorial disorder associated with considerable morbidity and mortality. The kidney plays a major role in the long term regulation of blood pressure. Liddle syndrome (pseudo-hyperaldosteronism) is one of a number of monogenic disorders of salt and water transport. In a kindred with at least four affected members suffering from Liddle syndrome, we confirmed by direct DNA sequencing the identity of a novel heterozygous mutation in h betaENaC, the gene encoding the beta subunit of the amiloride sensitive epithelial sodium channel which is expressed in the distal nephron. Single stranded conformational polymorphism analysis showed cosegregation of the mutant allele within the kindred with the Liddle phenotype. An insertion of an additional cytosine into a string of six located between codons 593 and 595 results in a sequence frameshift and is predicted to produce a protein truncated by 34 amino acids. The availability of a molecular diagnostic tool has implications for the management of hypertension and genetic counselling in families with Liddle syndrome.

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Year:  1998        PMID: 9643296      PMCID: PMC1051349          DOI: 10.1136/jmg.35.6.510

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  12 in total

1.  Mineralocorticoid action: target tissue specificity is enzyme, not receptor, mediated.

Authors:  J W Funder; P T Pearce; R Smith; A I Smith
Journal:  Science       Date:  1988-10-28       Impact factor: 47.728

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3.  Mechanism by which Liddle's syndrome mutations increase activity of a human epithelial Na+ channel.

Authors:  P M Snyder; M P Price; F J McDonald; C M Adams; K A Volk; B G Zeiher; J B Stokes; M J Welsh
Journal:  Cell       Date:  1995-12-15       Impact factor: 41.582

4.  Amiloride-sensitive epithelial Na+ channel is made of three homologous subunits.

Authors:  C M Canessa; L Schild; G Buell; B Thorens; I Gautschi; J D Horisberger; B C Rossier
Journal:  Nature       Date:  1994-02-03       Impact factor: 49.962

Review 5.  Science, medicine, and the future. Hypertension.

Authors:  M J Brown
Journal:  BMJ       Date:  1997-04-26

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Authors:  S Ulick; L S Levine; P Gunczler; G Zanconato; L C Ramirez; W Rauh; A Rösler; H L Bradlow; M I New
Journal:  J Clin Endocrinol Metab       Date:  1979-11       Impact factor: 5.958

7.  Hypertension caused by a truncated epithelial sodium channel gamma subunit: genetic heterogeneity of Liddle syndrome.

Authors:  J H Hansson; C Nelson-Williams; H Suzuki; L Schild; R Shimkets; Y Lu; C Canessa; T Iwasaki; B Rossier; R P Lifton
Journal:  Nat Genet       Date:  1995-09       Impact factor: 38.330

8.  Membrane topology of the amiloride-sensitive epithelial sodium channel.

Authors:  P M Snyder; F J McDonald; J B Stokes; M J Welsh
Journal:  J Biol Chem       Date:  1994-09-30       Impact factor: 5.157

9.  A chimaeric 11 beta-hydroxylase/aldosterone synthase gene causes glucocorticoid-remediable aldosteronism and human hypertension.

Authors:  R P Lifton; R G Dluhy; M Powers; G M Rich; S Cook; S Ulick; J M Lalouel
Journal:  Nature       Date:  1992-01-16       Impact factor: 49.962

10.  Liddle's syndrome: heritable human hypertension caused by mutations in the beta subunit of the epithelial sodium channel.

Authors:  R A Shimkets; D G Warnock; C M Bositis; C Nelson-Williams; J H Hansson; M Schambelan; J R Gill; S Ulick; R V Milora; J W Findling
Journal:  Cell       Date:  1994-11-04       Impact factor: 41.582

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  5 in total

Review 1.  Liddle syndrome in a Serbian family and literature review of underlying mutations.

Authors:  Radovan Bogdanović; Vladimir Kuburović; Nataša Stajić; Sadaf S Mughal; Alina Hilger; Sanja Ninić; Sergej Prijić; Michael Ludwig
Journal:  Eur J Pediatr       Date:  2011-09-29       Impact factor: 3.183

Review 2.  Hereditary causes of primary aldosteronism and other disorders of apparent excess mineralocorticoid activity.

Authors:  Xin He; Zubin Modi; Tobias Else
Journal:  Gland Surg       Date:  2020-02

3.  Phenotype-genotype analysis in two Chinese families with Liddle syndrome.

Authors:  Ling Gong; Jinxing Chen; Liying Shao; Weihua Song; Rutai Hui; Yibo Wang
Journal:  Mol Biol Rep       Date:  2014-01-29       Impact factor: 2.316

4.  Hypokalemia and hypertensive urgency in a 10-year-old boy: Answers.

Authors:  Ozlem Yuksel Aksoy; Funda Bastug; Binnaz Celik; Salih Uytun
Journal:  Pediatr Nephrol       Date:  2021-04-06       Impact factor: 3.714

Review 5.  Liddle Syndrome: Review of the Literature and Description of a New Case.

Authors:  Martina Tetti; Silvia Monticone; Jacopo Burrello; Patrizia Matarazzo; Franco Veglio; Barbara Pasini; Xavier Jeunemaitre; Paolo Mulatero
Journal:  Int J Mol Sci       Date:  2018-03-11       Impact factor: 5.923

  5 in total

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