Literature DB >> 8761527

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

D Lütjohann1, I Björkhem, L Ose.   

Abstract

Phytosterolaemia (sitosterolaemia) is a very rare inherited sterol storage disease characterized by tendon and tuberous xanthomas and by a predisposition to atherosclerosis. We here describe the first Scandinavian case. The 14-year-old female patient was found to have markedly elevated circulating levels of plant sterols (sitosterol, sitostanol, campesterol, stigmasterol), and the levels of these sterols were 20-50 times higher than in her healthy sister and heterozygous parents. In addition to the usual serum plant sterols we found a new major sterol in the patient tentatively identified as episterol or fecosterol (24-methyliden-cholest-7 (or 8)-en-3 beta-ol). A newly developed method based on the use of deuterium labelled cholesterol and plant sterols was used to measure sterol absorption in the patient and her relatives. Absorption of sitosterol averaged 20% in the patient and ranged from 4 to 8% in the relatives. Absorption of campesterol averaged 31% in the patient and ranged from 15 to 18% in her relatives. Absorption of cholesterol averaged 63% in the patient and ranged from 35 to 45% in the relatives. Cholesterol synthesis appeared to be reduced in the patient and was 46-52% of that of her relatives.

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Year:  1996        PMID: 8761527     DOI: 10.3109/00365519609088612

Source DB:  PubMed          Journal:  Scand J Clin Lab Invest        ISSN: 0036-5513            Impact factor:   1.713


  11 in total

1.  Fine mapping of a gene responsible for regulating dietary cholesterol absorption; founder effects underlie cases of phytosterolaemia in multiple communities.

Authors:  M H Lee; D Gordon; J Ott; K Lu; L Ose; T Miettinen; H Gylling; A F Stalenhoef; A Pandya; H Hidaka; B Brewer; H Kojima; N Sakuma; R Pegoraro; G Salen; S B Patel
Journal:  Eur J Hum Genet       Date:  2001-05       Impact factor: 4.246

Review 2.  Sitosterolemia--a rare disease. Are elevated plant sterols an additional risk factor?

Authors:  T Sudhop; K von Bergmann
Journal:  Z Kardiol       Date:  2004-12

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

Authors:  C Solcà; Z Stanga; B Pandit; P Diem; J Greeve; S B Patel
Journal:  Clin Genet       Date:  2005-08       Impact factor: 4.438

Review 4.  Genetic basis of sitosterolemia.

Authors:  M H Lee; K Lu; S B Patel
Journal:  Curr Opin Lipidol       Date:  2001-04       Impact factor: 4.776

Review 5.  Increased plasma levels of plant sterols and atherosclerosis: a controversial issue.

Authors:  Laura Calpe-Berdiel; Jesús Méndez-González; Francisco Blanco-Vaca; Joan Carles Escolà-Gil
Journal:  Curr Atheroscler Rep       Date:  2009-09       Impact factor: 5.113

6.  Comparison of the hepatic clearances of campesterol, sitosterol, and cholesterol in healthy subjects suggests that efflux transporters controlling intestinal sterol absorption also regulate biliary secretion.

Authors:  T Sudhop; Y Sahin; B Lindenthal; C Hahn; C Lüers; H K Berthold; K von Bergmann
Journal:  Gut       Date:  2002-12       Impact factor: 23.059

7.  Inhibition of cholesterol biosynthesis by Delta22-unsaturated phytosterols via competitive inhibition of sterol Delta24-reductase in mammalian cells.

Authors:  Carlos Fernández; Yajaira Suárez; Antonio J Ferruelo; Diego Gómez-Coronado; Miguel A Lasunción
Journal:  Biochem J       Date:  2002-08-15       Impact factor: 3.857

8.  Disruption of Abcg5 and Abcg8 in mice reveals their crucial role in biliary cholesterol secretion.

Authors:  Liqing Yu; Robert E Hammer; Jia Li-Hawkins; Klaus Von Bergmann; Dieter Lutjohann; Jonathan C Cohen; Helen H Hobbs
Journal:  Proc Natl Acad Sci U S A       Date:  2002-11-20       Impact factor: 11.205

Review 9.  Rediscovery of cerebrosterol.

Authors:  Ingemar Björkhem
Journal:  Lipids       Date:  2007-01-23       Impact factor: 1.880

Review 10.  Sitosterolemia: Twenty Years of Discovery of the Function of ABCG5ABCG8.

Authors:  Kori Williams; Allison Segard; Gregory A Graf
Journal:  Int J Mol Sci       Date:  2021-03-05       Impact factor: 5.923

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