Literature DB >> 15015036

Screening for point mutations in the LDL receptor gene in Bulgarian patients with severe hypercholesterolemia.

Vassil A Mihaylov1, Anelia D Horvath1, Alexey S Savov2, Elina F Kurshelova3, Ivanka D Paskaleva4, Assen R Goudev3, Ivaylo R Stoilov5, Varban S Ganev6.   

Abstract

Familial hypercholesterolemia (FH) is a common, autosomal dominant disorder of lipid metabolism, caused by defects in the receptor-mediated uptake of LDL (low-density lipoproteins) due to mutations in the LDL receptor gene ( LDLR). Mutations underlying FH in Bulgaria are largely unknown. The aim of the present study was to provide information about the spectrum of point mutations in LDLR in a sample of 45 Bulgarian patients with severe hypercholesterolemia. Exons 3, 4, 6, 8, 9, and 14, previously shown to be mutational hot spots in LDLR, were screened using PCR-single-strand conformation polymorphism (SSCP). Samples with abnormal SSCP patterns were sequenced. Three different, hitherto undescribed point mutations (367T>A, 377T>A, 917C>A) and two previously described mutations (858C>A and 1301C>T) in eight unrelated patients were identified; four of the detected point mutations being missense mutations and one, a nonsense mutation. One of the newly described point mutations (917C>A) is a base substitution at a nucleotide position, at which two other different base substitutions have already been reported. Thus, all three possible base substitutions at this nucleotide position have been detected, making it a hot spot for point mutations causing FH. This is the first such mutational hot spot described in exon 6 of LDLR.

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Year:  2004        PMID: 15015036     DOI: 10.1007/s10038-004-0127-6

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  26 in total

1.  High sensitivity of the single-strand conformation polymorphism method for detecting sequence variations in the low-density lipoprotein receptor gene validated by DNA sequencing.

Authors:  H K Jensen; L G Jensen; P S Hansen; O Faergeman; N Gregersen
Journal:  Clin Chem       Date:  1996-08       Impact factor: 8.327

2.  Low density lipoprotein receptor (LDLR) gene mutations in Canadian subjects with familial hypercholesterolemia, but not of French descent.

Authors:  J Wang; E Huff; L Janecka; R A Hegele
Journal:  Hum Mutat       Date:  2001-10       Impact factor: 4.878

3.  Spectrum of low density lipoprotein receptor mutations in Czech hypercholesterolemic patients.

Authors:  Viera Kuhrová; Hana Francová; Petra Zapletalová; Tomás Freiberger; Lenka Fajkusová; Eva Hrabincová; Romana Slováĉková; Libor Kozák; Romana Slováková
Journal:  Hum Mutat       Date:  2002-01       Impact factor: 4.878

4.  Detection of polymorphisms of human DNA by gel electrophoresis as single-strand conformation polymorphisms.

Authors:  M Orita; H Iwahana; H Kanazawa; K Hayashi; T Sekiya
Journal:  Proc Natl Acad Sci U S A       Date:  1989-04       Impact factor: 11.205

5.  Heterogeneity of mutations in Bulgarian phenylketonuria haplotype 1 and 4 alleles.

Authors:  L Kalaydjieva; B Dworniczak; I Kremensky; K Koprivarova; B Radeva; R Milusheva; C Aulehla-Scholz; J Horst
Journal:  Clin Genet       Date:  1992-03       Impact factor: 4.438

6.  Influence of genotype at the low density lipoprotein (LDL) receptor gene locus on the clinical phenotype and response to lipid-lowering drug therapy in heterozygous familial hypercholesterolaemia. The Familial Hypercholesterolaemia Regression Study Group.

Authors:  X M Sun; D D Patel; B L Knight; A K Soutar
Journal:  Atherosclerosis       Date:  1998-01       Impact factor: 5.162

7.  Characterization of mutations in the low density lipoprotein (LDL)-receptor gene in patients with homozygous familial hypercholesterolemia, and frequency of these mutations in FH patients in the United Kingdom.

Authors:  J C Webb; X M Sun; S N McCarthy; C Neuwirth; G R Thompson; B L Knight; A K Soutar
Journal:  J Lipid Res       Date:  1996-02       Impact factor: 5.922

8.  Genetic determinants of responsiveness to the HMG-CoA reductase inhibitor fluvastatin in patients with molecularly defined heterozygous familial hypercholesterolemia.

Authors:  E Leitersdorf; S Eisenberg; O Eliav; Y Friedlander; N Berkman; E J Dann; D Landsberger; E Sehayek; V Meiner; M Wurm
Journal:  Circulation       Date:  1993-04       Impact factor: 29.690

Review 9.  Molecular genetics of the LDL receptor gene in familial hypercholesterolemia.

Authors:  H H Hobbs; M S Brown; J L Goldstein
Journal:  Hum Mutat       Date:  1992       Impact factor: 4.878

10.  Association of specific LDL receptor gene mutations with differential plasma lipoprotein response to simvastatin in young French Canadians with heterozygous familial hypercholesterolemia.

Authors:  P Couture; L D Brun; F Szots; M Lelièvre; D Gaudet; J P Després; J Simard; P J Lupien; C Gagné
Journal:  Arterioscler Thromb Vasc Biol       Date:  1998-06       Impact factor: 8.311

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  1 in total

1.  Familial hypercholesterolemia in St-Petersburg: the known and novel mutations found in the low density lipoprotein receptor gene in Russia.

Authors:  Faina M Zakharova; Dorte Damgaard; Michail Y Mandelshtam; Valery I Golubkov; Peter H Nissen; Gitte G Nilsen; Anette Stenderup; Boris M Lipovetsky; Vladimir O Konstantinov; Alexander D Denisenko; Vadim B Vasilyev; Ole Faergeman
Journal:  BMC Med Genet       Date:  2005-02-08       Impact factor: 2.103

  1 in total

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