Literature DB >> 8900229

Novel molecular variants of the Na-Cl cotransporter gene are responsible for Gitelman syndrome.

N Mastroianni1, A Bettinelli, M Bianchetti, G Colussi, M De Fusco, F Sereni, A Ballabio, G Casari.   

Abstract

A hereditary defect of the distal tubule accounts for the clinical features of Gitelman syndrome (GS), an autosomal recessive disease characterized by hypokalemia, hypomagnesemia, metabolic alkalosis, and hypocalciuria. Recently, we cloned the cDNA coding for the human Na-Cl thiazide-sensitive cotransporter (TSC; also known as ¿NCCT¿ or ¿SLC12A3¿) as a possible candidate for GS, and Simon et al., independently, described mutations in patients with GS. Now, we show 12 additional mutations consistent with a loss of function of the Na-Cl cotransporter in GS. Two missense replacements, R209W and P349L, are common to both studies and could represent ancient mutations. The other mutations include three deletions, two insertions, and six missense mutations. When all mutations from both studies are considered, missense mutations seem to be more frequently localized within the intracellular domains of the molecule, rather than in transmembrane or extracellular domains. One family, previously reported as a GS form with dominant inheritance, has proved to be recessive, with the affected child being a compound heterozygote. A highly informative intragenic tetranucleotide marker, useful for molecular diagnostic studies, has been identified at the acceptor splice site of exon 9.

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Year:  1996        PMID: 8900229      PMCID: PMC1914834     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  12 in total

1.  The skeletal muscle chloride channel in dominant and recessive human myotonia.

Authors:  M C Koch; K Steinmeyer; C Lorenz; K Ricker; F Wolf; M Otto; B Zoll; F Lehmann-Horn; K H Grzeschik; T J Jentsch
Journal:  Science       Date:  1992-08-07       Impact factor: 47.728

2.  Bartter's syndrome: evidence suggesting a distal tubular defect in a hypocalciuric variant of the syndrome.

Authors:  R A Sutton; V Mavichak; A Halabe; G E Wilkins
Journal:  Miner Electrolyte Metab       Date:  1992

3.  Rapid and sensitive detection of point mutations and DNA polymorphisms using the polymerase chain reaction.

Authors:  M Orita; Y Suzuki; T Sekiya; K Hayashi
Journal:  Genomics       Date:  1989-11       Impact factor: 5.736

4.  Shaker K+ channel T1 domain self-tetramerizes to a stable structure.

Authors:  P J Pfaffinger; D DeRubeis
Journal:  J Biol Chem       Date:  1995-12-01       Impact factor: 5.157

5.  A new familial disorder characterized by hypokalemia and hypomagnesemia.

Authors:  H J Gitelman; J B Graham; L G Welt
Journal:  Trans Assoc Am Physicians       Date:  1966

6.  Calcium metabolism and calciotropic hormone levels in Gitelman's syndrome.

Authors:  G Colussi; M Macaluso; C Brunati; L Minetti
Journal:  Miner Electrolyte Metab       Date:  1994

7.  Acute intermittent porphyria in Finland: 19 mutations in the porphobilinogen deaminase gene.

Authors:  R Kauppinen; S Mustajoki; H Pihlaja; L Peltonen; P Mustajoki
Journal:  Hum Mol Genet       Date:  1995-02       Impact factor: 6.150

8.  Genetic heterogeneity in tubular hypomagnesemia-hypokalemia with hypocalcuria (Gitelman's syndrome).

Authors:  A Bettinelli; M G Bianchetti; P Borella; E Volpini; M G Metta; E Basilico; A Selicorni; A Bargellini; M R Grassi
Journal:  Kidney Int       Date:  1995-02       Impact factor: 10.612

9.  Use of calcium excretion values to distinguish two forms of primary renal tubular hypokalemic alkalosis: Bartter and Gitelman syndromes.

Authors:  A Bettinelli; M G Bianchetti; E Girardin; A Caringella; M Cecconi; A C Appiani; L Pavanello; R Gastaldi; C Isimbaldi; G Lama
Journal:  J Pediatr       Date:  1992-01       Impact factor: 4.406

10.  Molecular cloning, expression pattern, and chromosomal localization of the human Na-Cl thiazide-sensitive cotransporter (SLC12A3).

Authors:  N Mastroianni; M De Fusco; M Zollo; G Arrigo; O Zuffardi; A Bettinelli; A Ballabio; G Casari
Journal:  Genomics       Date:  1996-08-01       Impact factor: 5.736

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  36 in total

Review 1.  Molecular physiology of cation-coupled Cl- cotransport: the SLC12 family.

Authors:  Steven C Hebert; David B Mount; Gerardo Gamba
Journal:  Pflugers Arch       Date:  2003-05-09       Impact factor: 3.657

2.  Novel NCC mutants and functional analysis in a new cohort of patients with Gitelman syndrome.

Authors:  Bob Glaudemans; Helger G Yntema; Pedro San-Cristobal; Jeroen Schoots; Rolph Pfundt; Erik-J Kamsteeg; René J Bindels; Nine V A M Knoers; Joost G Hoenderop; Lies H Hoefsloot
Journal:  Eur J Hum Genet       Date:  2011-10-19       Impact factor: 4.246

Review 3.  WNK kinases, renal ion transport and hypertension.

Authors:  Pedro San-Cristobal; Paola de los Heros; José Ponce-Coria; Erika Moreno; Gerardo Gamba
Journal:  Am J Nephrol       Date:  2008-06-12       Impact factor: 3.754

Review 4.  The biochemical diagnosis of Gitelman disease and the definition of "hypocalciuria".

Authors:  Mario G Bianchetti; Alberto Edefonti; Alberto Bettinelli
Journal:  Pediatr Nephrol       Date:  2003-05       Impact factor: 3.714

5.  Phosphorylation decreases ubiquitylation of the thiazide-sensitive cotransporter NCC and subsequent clathrin-mediated endocytosis.

Authors:  Lena L Rosenbaek; Marleen L A Kortenoeven; Takwa S Aroankins; Robert A Fenton
Journal:  J Biol Chem       Date:  2014-03-25       Impact factor: 5.157

6.  A labor- and cost-effective non-optical semiconductor (Ion Torrent) next-generation sequencing of the SLC12A3 and CLCNKA/B genes in Gitelman's syndrome patients.

Authors:  Beatriz Tavira; Juan Gómez; Fernando Santos; Helena Gil; Victoria Alvarez; Eliecer Coto
Journal:  J Hum Genet       Date:  2014-05-15       Impact factor: 3.172

7.  Recurrent deep intronic mutations in the SLC12A3 gene responsible for Gitelman's syndrome.

Authors:  Yi-Fen Lo; Kandai Nozu; Kazumoto Iijima; Takahiro Morishita; Che-Chung Huang; Sung-Sen Yang; Huey-Kang Sytwu; Yu-Wei Fang; Min-Hua Tseng; Shih-Hua Lin
Journal:  Clin J Am Soc Nephrol       Date:  2010-11-04       Impact factor: 8.237

Review 8.  Physiology of SLC12 transporters: lessons from inherited human genetic mutations and genetically engineered mouse knockouts.

Authors:  Kenneth B Gagnon; Eric Delpire
Journal:  Am J Physiol Cell Physiol       Date:  2013-01-16       Impact factor: 4.249

Review 9.  Molecular determinants of magnesium homeostasis: insights from human disease.

Authors:  R Todd Alexander; Joost G Hoenderop; René J Bindels
Journal:  J Am Soc Nephrol       Date:  2008-06-18       Impact factor: 10.121

10.  Characterization of a novel phosphorylation site in the sodium-chloride cotransporter, NCC.

Authors:  L L Rosenbaek; M Assentoft; N B Pedersen; N MacAulay; R A Fenton
Journal:  J Physiol       Date:  2012-09-10       Impact factor: 5.182

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