Literature DB >> 3633381

Detection of familial hypercholesterolemia by assaying functional low-density-lipoprotein receptors on lymphocytes.

J A Cuthbert, C A East, D W Bilheimer, P E Lipsky.   

Abstract

In familial hypercholesterolemia, structural and functional abnormalities of the receptor for low-density lipoprotein (LDL) lead to hypercholesterolemia and premature atherosclerosis. We have developed a simplified method to identify LDL-receptor defects in peripheral-blood lymphocytes. When lymphocytes are cultured in lipoprotein-depleted medium and endogenous sterol biosynthesis is suppressed with mevinolin, mitogen-stimulated proliferation of lymphocytes is dependent on an exogenous source of cholesterol. Whereas a small concentration of supplemental LDL cholesterol (3 to 4 micrograms per milliliter) permits a maximal response in normal lymphocytes, even high concentrations (10 to 50 micrograms per milliliter) are unable to support the proliferation of lymphocytes from patients with homozygous familial hypercholesterolemia. Thus, functional LDL receptors are necessary to allow lymphocyte proliferation in these cultures. The response of lymphocytes from patients with hyperlipidemia not caused by defective LDL receptors was like that of normal cells. In contrast, the response of lymphocytes from patients with heterozygous familial hypercholesterolemia was intermediate between that of homozygotes and that of normal or hyperlipidemic controls. Our method can therefore be used to identify persons who are heterozygous for abnormalities of LDL receptors.

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Year:  1986        PMID: 3633381     DOI: 10.1056/NEJM198604033141404

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  19 in total

1.  Clinically used selective oestrogen receptor modulators increase LDL receptor activity in primary human lymphocytes.

Authors:  F Cerrato; M E Fernández-Suárez; R Alonso; M Alonso; C Vázquez; O Pastor; P Mata; M A Lasunción; D Gómez-Coronado
Journal:  Br J Pharmacol       Date:  2015-01-08       Impact factor: 8.739

2.  Combined analysis of genome scans of dutch and finnish families reveals a susceptibility locus for high-density lipoprotein cholesterol on chromosome 16q.

Authors:  Päivi Pajukanta; Hooman Allayee; Kelly L Krass; Ali Kuraishy; Aino Soro; Heidi E Lilja; Rebecca Mar; Marja-Riitta Taskinen; Ilpo Nuotio; Markku Laakso; Jerome I Rotter; Tjerk W A de Bruin; Rita M Cantor; Aldons J Lusis; Leena Peltonen
Journal:  Am J Hum Genet       Date:  2003-03-12       Impact factor: 11.025

3.  Genomewide scan for familial combined hyperlipidemia genes in finnish families, suggesting multiple susceptibility loci influencing triglyceride, cholesterol, and apolipoprotein B levels.

Authors:  P Pajukanta; J D Terwilliger; M Perola; T Hiekkalinna; I Nuotio; P Ellonen; M Parkkonen; J Hartiala; K Ylitalo; J Pihlajamäki; K Porkka; M Laakso; J Viikari; C Ehnholm; M R Taskinen; L Peltonen
Journal:  Am J Hum Genet       Date:  1999-05       Impact factor: 11.025

4.  The prediction of the therapeutic response to cholesterol lowering drugs in an 11-year-old boy with homozygous familial hypercholesterolaemia.

Authors:  H D Bakker; T Bruin; M C Schaap; P J Lansberg; J J Kastelein
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

5.  Pharmacokinetics and response to pravastatin in paediatric patients with familial hypercholesterolaemia and in paediatric cardiac transplant recipients in relation to polymorphisms of the SLCO1B1 and ABCB1 genes.

Authors:  Mia Hedman; Marjatta Antikainen; Christer Holmberg; Mikko Neuvonen; Michel Eichelbaum; Kari T Kivistö; Pertti J Neuvonen; Mikko Niemi
Journal:  Br J Clin Pharmacol       Date:  2006-06       Impact factor: 4.335

Review 6.  Mechanism of action and biological profile of HMG CoA reductase inhibitors. A new therapeutic alternative.

Authors:  E E Slater; J S MacDonald
Journal:  Drugs       Date:  1988       Impact factor: 9.546

7.  Teaching self-catheterization skills to children with neurogenic bladder complications.

Authors:  N A Neef; J M Parrish; K F Hannigan; T J Page; B A Iwata
Journal:  J Appl Behav Anal       Date:  1989

8.  An improved method on stimulated T-lymphocytes to functionally characterize novel and known LDLR mutations.

Authors:  Maria Romano; Maria Donata Di Taranto; Peppino Mirabelli; Maria Nicoletta D'Agostino; Arcangelo Iannuzzi; Gennaro Marotta; Marco Gentile; Maddalena Raia; Rosa Di Noto; Luigi Del Vecchio; Paolo Rubba; Giuliana Fortunato
Journal:  J Lipid Res       Date:  2011-08-24       Impact factor: 5.922

9.  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

10.  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

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