Literature DB >> 1319734

Characterization of deletions in the LDL receptor gene in patients with familial hypercholesterolemia in the United Kingdom.

X M Sun1, J C Webb, V Gudnason, S Humphries, M Seed, G R Thompson, B L Knight, A K Soutar.   

Abstract

A sample of 200 patients with a clinical diagnosis of heterozygous (189) or homozygous (11) familial hypercholesterolemia (FH) attending lipid clinics in the London area have been screened for the presence of major gene defects in the low density lipoprotein (LDL) receptor gene by Southern blotting of genomic DNA with specific probes. This study is part of a project to determine the frequency of known mutations in the LDL receptor gene in this population. A new polymorphism for the enzyme Bgl II was identified by hybridization with a probe specific for the promoter plus exon 1 of the LDL receptor gene. The observed frequency of the rare allele, characterized by a Bgl II fragment of 13 kb compared with 10 kb for the common allele, was 0.08 in this group of FH patients. Several individuals who were heterozygous for the rare allele were also heterozygous for a mutation elsewhere in the LDL receptor gene that is known to cause FH. Eight different mutations, seven deletions and one duplication, were detected in a total of nine patients, accounting for 4.5% of the mutant alleles in this group. Three of the mutations are apparently identical to deletions that have been described previously in FH patients of British or European origin, while the remaining five have not been described. Two of these were in patients of Polish and Asian Indian origin, while the other three were in patients of apparently British ancestry.

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Year:  1992        PMID: 1319734     DOI: 10.1161/01.atv.12.7.762

Source DB:  PubMed          Journal:  Arterioscler Thromb        ISSN: 1049-8834


  16 in total

1.  Identification of a common low density lipoprotein receptor mutation (R329X) in the south of England: complete linkage disequilibrium with an allele of microsatellite D19S394.

Authors:  I N Day; L Haddad; S D O'Dell; L B Day; R A Whittall; S E Humphries
Journal:  J Med Genet       Date:  1997-02       Impact factor: 6.318

2.  Universal primer quantitative fluorescent multiplex (UPQFM) PCR: a method to detect major and minor rearrangements of the low density lipoprotein receptor gene.

Authors:  K E Heath; I N Day; S E Humphries
Journal:  J Med Genet       Date:  2000-04       Impact factor: 6.318

3.  1992 American Society of Human Genetics presidential address: back to the future.

Authors:  W E Nance
Journal:  Am J Hum Genet       Date:  1993-07       Impact factor: 11.025

4.  Mutational analysis in UK patients with a clinical diagnosis of familial hypercholesterolaemia: relationship with plasma lipid traits, heart disease risk and utility in relative tracing.

Authors:  Steve E Humphries; Treena Cranston; Marcus Allen; Helen Middleton-Price; Maryam C Fernandez; Victoria Senior; Emma Hawe; Andrew Iversen; Richard Wray; Martin A Crook; Anthony S Wierzbicki
Journal:  J Mol Med (Berl)       Date:  2005-12-31       Impact factor: 4.599

5.  Characterization of a novel cellular defect in patients with phenotypic homozygous familial hypercholesterolemia.

Authors:  D Norman; X M Sun; M Bourbon; B L Knight; R P Naoumova; A K Soutar
Journal:  J Clin Invest       Date:  1999-09       Impact factor: 14.808

6.  Use of the single-strand conformational polymorphism method to detect recurrent and novel mutations in the low-density lipoprotein receptor gene in patients with familial hypercholesterolaemia: detection of a novel mutation Asp200-->Gly.

Authors:  V Gudnason; Y T Mak; J Betteridge; S N McCarthy; S Humphries
Journal:  Clin Investig       Date:  1993-04

7.  Screening for mutations in exon 4 of the LDL receptor gene in a German population with severe hypercholesterolemia.

Authors:  J Giesel; G Holzem; K Oette
Journal:  Hum Genet       Date:  1995-09       Impact factor: 4.132

8.  Six DNA polymorphisms in the low density lipoprotein receptor gene: their genetic relationship and an example of their use for identifying affected relatives of patients with familial hypercholesterolaemia.

Authors:  S Humphries; L King-Underwood; V Gudnason; M Seed; S Delattre; V Clavey; J C Fruchart
Journal:  J Med Genet       Date:  1993-04       Impact factor: 6.318

9.  The role of calcium in lipoprotein release by the low-density lipoprotein receptor.

Authors:  Zhenze Zhao; Peter Michaely
Journal:  Biochemistry       Date:  2009-08-04       Impact factor: 3.162

10.  Identification of a common low density lipoprotein receptor mutation (C163Y) in the west of Scotland.

Authors:  W K Lee; L Haddad; M J Macleod; A M Dorrance; D J Wilson; D Gaffney; M H Dominiczak; C J Packard; I N Day; S E Humphries; A F Dominiczak
Journal:  J Med Genet       Date:  1998-07       Impact factor: 6.318

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