Literature DB >> 2760198

Finnish type of low density lipoprotein receptor gene mutation (FH-Helsinki) deletes exons encoding the carboxy-terminal part of the receptor and creates an internalization-defective phenotype.

K Aalto-Setälä1, E Helve, P T Kovanen, K Kontula.   

Abstract

A specific type of gene mutation affecting the LDL receptor has been found in many Finnish patients with familial hypercholesterolemia (FH). The mutant allele is characterized by a 9.5-kb deletion extending from intron 15 to exon 18. Molecular cloning and sequencing of a cDNA segment corresponding to the deleted allele indicated that the mutant receptor differs radically from the normal one because of loss of the domains encoded by exons 16, 17, and 18. The carboxy-terminal portion of the normal receptor, comprising the amino acids 750-839, has been replaced by an unrelated stretch of 55 amino acids. The mutant allele was found to occur in 23 (50%) of 46 unrelated FH patients with an established functional defect in the LDL receptor. In cultured fibroblasts from the FH patients with the 9.5-kb deletion, both receptor-mediated binding and internalization of 125I-LDL were lower than normal, the former, on average, by 25%, and the latter, on average, by 50%. This combined functional defect probably results from both impaired attachment and impaired internalization of the mutated receptor. It remains to be investigated whether this Finnish type of LDL receptor gene mutation, here designated FH-Helsinki, occurs in other ethnic groups.

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Year:  1989        PMID: 2760198      PMCID: PMC548909          DOI: 10.1172/JCI114192

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  34 in total

1.  The low density lipoprotein receptor. Identification of amino acids in cytoplasmic domain required for rapid endocytosis.

Authors:  C G Davis; I R van Driel; D W Russell; M S Brown; J L Goldstein
Journal:  J Biol Chem       Date:  1987-03-25       Impact factor: 5.157

Review 2.  Hereditary diseases in Finland; rare flora in rare soul.

Authors:  R Norio; H R Nevanlinna; J Perheentupa
Journal:  Ann Clin Res       Date:  1973-06

3.  The metabolism of very low density lipoprotein proteins. I. Preliminary in vitro and in vivo observations.

Authors:  D W Bilheimer; S Eisenberg; R I Levy
Journal:  Biochim Biophys Acta       Date:  1972-02-21

4.  Deletion of exon encoding cysteine-rich repeat of low density lipoprotein receptor alters its binding specificity in a subject with familial hypercholesterolemia.

Authors:  H H Hobbs; M S Brown; J L Goldstein; D W Russell
Journal:  J Biol Chem       Date:  1986-10-05       Impact factor: 5.157

5.  The Lebanese allele at the low density lipoprotein receptor locus. Nonsense mutation produces truncated receptor that is retained in endoplasmic reticulum.

Authors:  M A Lehrman; W J Schneider; M S Brown; C G Davis; A Elhammer; D W Russell; J L Goldstein
Journal:  J Biol Chem       Date:  1987-01-05       Impact factor: 5.157

6.  Unequal crossing-over between two alu-repetitive DNA sequences in the low-density-lipoprotein-receptor gene. A possible mechanism for the defect in a patient with familial hypercholesterolaemia.

Authors:  B Horsthemke; U Beisiegel; A Dunning; J R Havinga; R Williamson; S Humphries
Journal:  Eur J Biochem       Date:  1987-04-01

7.  Alu-Alu recombination deletes splice acceptor sites and produces secreted low density lipoprotein receptor in a subject with familial hypercholesterolemia.

Authors:  M A Lehrman; D W Russell; J L Goldstein; M S Brown
Journal:  J Biol Chem       Date:  1987-03-05       Impact factor: 5.157

8.  Familial hypercholesterolemia in South African Afrikaners. PvuII and StuI DNA polymorphisms in the LDL-receptor gene consistent with a predominating founder gene effect.

Authors:  P A Brink; L T Steyn; G A Coetzee; D R Van der Westhuyzen
Journal:  Hum Genet       Date:  1987-09       Impact factor: 4.132

9.  Binding and degradation of low density lipoproteins by cultured human fibroblasts. Comparison of cells from a normal subject and from a patient with homozygous familial hypercholesterolemia.

Authors:  J L Goldstein; M S Brown
Journal:  J Biol Chem       Date:  1974-08-25       Impact factor: 5.157

10.  Identification of deletions in the human low density lipoprotein receptor gene.

Authors:  B Horsthemke; A Dunning; S Humphries
Journal:  J Med Genet       Date:  1987-03       Impact factor: 6.318

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  18 in total

1.  Use of DNA haplotype analysis in diagnosis of familial hypercholesterolaemia in 31 German families.

Authors:  H Schuster; G Rauh; C Gerl; C Keller; G Wolfram; N Zöllner
Journal:  J Med Genet       Date:  1991-12       Impact factor: 6.318

2.  Screening for a prevalent LDL receptor mutation in patients with severe hypercholesterolaemia.

Authors:  M J Savolainen; T Korhonen; K Aalto-Setälä; K Kontula; Y A Kesäniemi
Journal:  Hum Genet       Date:  1991-06       Impact factor: 4.132

3.  The familial hypercholesterolemia (FH)-North Karelia mutation of the low density lipoprotein receptor gene deletes seven nucleotides of exon 6 and is a common cause of FH in Finland.

Authors:  U M Koivisto; H Turtola; K Aalto-Setälä; B Top; R R Frants; P T Kovanen; A C Syvänen; K Kontula
Journal:  J Clin Invest       Date:  1992-07       Impact factor: 14.808

4.  A missense mutation in the low density lipoprotein receptor gene causes familial hypercholesterolemia in Sephardic Jews.

Authors:  E Leitersdorf; A Reshef; V Meiner; E J Dann; Y Beigel; F G van Roggen; D R van der Westhuyzen; G A Coetzee
Journal:  Hum Genet       Date:  1993-03       Impact factor: 4.132

5.  Hypercholesterolemia in five Israeli Christian-Arab kindreds is caused by the "Lebanese" allele at the low density lipoprotein receptor gene locus and by an additional independent major factor.

Authors:  A Oppenheim; Y Friedlander; E J Dann; N Berkman; S P Schwartz; E Leitersdorf
Journal:  Hum Genet       Date:  1991-11       Impact factor: 4.132

6.  Polymorphic haplotypes and recombination rates at the LDL receptor gene locus in subjects with and without familial hypercholesterolemia who are from different populations.

Authors:  A R Miserez; H Schuster; N Chiodetti; U Keller
Journal:  Am J Hum Genet       Date:  1993-04       Impact factor: 11.025

Review 7.  Apolipoprotein genes and atherosclerosis.

Authors:  J L Breslow
Journal:  Clin Investig       Date:  1992-05

8.  Low density lipoprotein receptor founder mutations in Afrikaner familial hypercholesterolaemic patients: a comparison of two geographical areas.

Authors:  F Graadt van Roggen; D R van der Westhuyzen; A D Marais; W Gevers; G A Coetzee
Journal:  Hum Genet       Date:  1991-12       Impact factor: 4.132

9.  Molecular characterization of minor gene rearrangements in Finnish patients with heterozygous familial hypercholesterolemia: identification of two common missense mutations (Gly823-->Asp and Leu380-->His) and eight rare mutations of the LDL receptor gene.

Authors:  U M Koivisto; J S Viikari; K Kontula
Journal:  Am J Hum Genet       Date:  1995-10       Impact factor: 11.025

10.  Screening for mutations in exon 4 of the LDL receptor gene: identification of a new deletion mutation.

Authors:  L Theart; M J Kotze; E Langenhoven; O Loubser; A V Peeters; C J Lintott; R S Scott
Journal:  J Med Genet       Date:  1995-05       Impact factor: 6.318

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