Literature DB >> 15996216

Sitosterolaemia in Switzerland: molecular genetics links the US Amish-Mennonites to their European roots.

C Solcà1, Z Stanga, B Pandit, P Diem, J Greeve, S B Patel.   

Abstract

Sitosterolaemia is a rare autosomal recessive disease characterized by increased intestinal absorption of plant sterols, decreased hepatic excretion into bile and elevated concentrations in plasma phytosterols. Homozygous or compound heterozygous loss of function mutations in either of the ATP-binding cassette (ABC) proteins ABCG5 and ABCG8 explain the increased absorption of plant sterols. Here we report a Swiss index patient with sitosterolaemia, who presented with the classical symptoms of xanthomas, but also had mitral and aortic valvular heart disease. Her management over the last 20 years included a novel therapeutic approach of high-dose cholesterol feeding that was semi-effective. Mutational and extended haplotype analyses showed that our patient shared this haplotype with that of the Amish-Mennonite sitosterolaemia patients, indicating they are related ancestrally.

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Year:  2005        PMID: 15996216      PMCID: PMC1201539          DOI: 10.1111/j.1399-0004.2005.00472.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  35 in total

1.  Increased sitosterol absorption is offset by rapid elimination to prevent accumulation in heterozygotes with sitosterolemia.

Authors:  G Salen; G S Tint; S Shefer; V Shore; L Nguyen
Journal:  Arterioscler Thromb       Date:  1992-05

2.  Unexpected failure of bile acid malabsorption to stimulate cholesterol synthesis in sitosterolemia with xanthomatosis. Comparison with lovastatin.

Authors:  L Nguyen; G Salen; S Shefer; V Shore; G S Tint; G Ness
Journal:  Arteriosclerosis       Date:  1990 Mar-Apr

3.  Phytosterolaemia in a Norwegian family: diagnosis and characterization of the first Scandinavian case.

Authors:  D Lütjohann; I Björkhem; L Ose
Journal:  Scand J Clin Lab Invest       Date:  1996-05       Impact factor: 1.713

4.  Response to diet and cholestyramine in a patient with sitosterolemia.

Authors:  P F Belamarich; R J Deckelbaum; T J Starc; B E Dobrin; G S Tint; G Salen
Journal:  Pediatrics       Date:  1990-12       Impact factor: 7.124

5.  In vivo metabolism-based discovery of a potent cholesterol absorption inhibitor, SCH58235, in the rat and rhesus monkey through the identification of the active metabolites of SCH48461.

Authors:  M Van Heek; C F France; D S Compton; R L McLeod; N P Yumibe; K B Alton; E J Sybertz; H R Davis
Journal:  J Pharmacol Exp Ther       Date:  1997-10       Impact factor: 4.030

6.  Sterol absorption and sterol balance in phytosterolemia evaluated by deuterium-labeled sterols: effect of sitostanol treatment.

Authors:  D Lütjohann; I Björkhem; U F Beil; K von Bergmann
Journal:  J Lipid Res       Date:  1995-08       Impact factor: 5.922

Review 7.  Sitosterolemia.

Authors:  G Salen; S Shefer; L Nguyen; G C Ness; G S Tint; V Shore
Journal:  J Lipid Res       Date:  1992-07       Impact factor: 5.922

8.  Sluggish sitosterol turnover and hepatic failure to excrete sitosterol into bile cause expansion of body pool of sitosterol in patients with sitosterolemia and xanthomatosis.

Authors:  A K Bhattacharyya; W E Connor; D S Lin; M M McMurry; R S Shulman
Journal:  Arterioscler Thromb       Date:  1991 Sep-Oct

9.  Sitosterolemia: opposing effects of cholestyramine and lovastatin on plasma sterol levels in a homozygous girl and her heterozygous father.

Authors:  M M Cobb; G Salen; G S Tint; J Greenspan; L B Nguyen
Journal:  Metabolism       Date:  1996-06       Impact factor: 8.694

10.  Regulation of cholesterol biosynthesis in sitosterolemia: effects of lovastatin, cholestyramine, and dietary sterol restriction.

Authors:  L B Nguyen; M Cobb; S Shefer; G Salen; G C Ness; G S Tint
Journal:  J Lipid Res       Date:  1991-12       Impact factor: 5.922

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  3 in total

1.  Phytosterols and phytosterolemia: gene-diet interactions.

Authors:  Maria C Izar; Daniela M Tegani; Soraia H Kasmas; Francisco A Fonseca
Journal:  Genes Nutr       Date:  2010-08-28       Impact factor: 5.523

2.  The ABCG8 G574R variant, serum plant sterol levels, and cardiovascular disease risk in the Old Order Amish.

Authors:  Richard B Horenstein; Braxton D Mitchell; Wendy S Post; Dieter Lütjohann; Klaus von Bergmann; Kathleen A Ryan; Michael Terrin; Alan R Shuldiner; Nanette I Steinle
Journal:  Arterioscler Thromb Vasc Biol       Date:  2012-12-13       Impact factor: 8.311

Review 3.  Sitosterolemia: diagnosis, investigation, and management.

Authors:  Joan Carles Escolà-Gil; Helena Quesada; Josep Julve; Jesús M Martín-Campos; Lídia Cedó; Francisco Blanco-Vaca
Journal:  Curr Atheroscler Rep       Date:  2014-07       Impact factor: 5.113

  3 in total

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