Literature DB >> 29055934

Cryptogenic Cirrhosis and Sitosterolemia: A Treatable Disease If Identified but Fatal If Missed.

Fateh Bazerbachi1, Erin E Conboy2, Taofic Mounajjed3, Kymberly D Watt1, Dusica Babovic-Vuksanovic2, Shailendra B Patel4, Patrick S Kamath1.   

Abstract

Sitosterolemia is an autosomal recessive metabolic disease caused by mutations in ABCG5 or ABCG8 genes which encode for the (ATP)-binding cassette (ABC) transporters that are responsible for the trafficking of xenosterols. Liver involvement is not a recognized manifestation of this disease, and cirrhosis has been reported only once in the medical literature. We describe a fatal case of a 21-year old South Asian male who presented with decompensated cirrhosis, and biochemical abnormalities consistent with sitosterolemia. Genetic testing showed a homozygous pathogenic mutation in ABCG5, confirming the diagnosis. Sitosterolemia is a rare, but likely under-recognized condition, and a high degree of suspicion is imperative to make the diagnosis. We propose that sitosterolemia should be included in the differential diagnosis for patients with cryptogenic cirrhosis, especially as there are effective oral therapies to treat this condition. Newly diagnosed sitosterolemia patients should undergo a thorough hepatology evaluation and follow-up to evaluate for the presence, development, and progression of any hepatic involvement.

Entities:  

Keywords:  Cryptogenic cirrhosis. Ascites. Idiopathic cirrhosis. Sitosterolemia. Phytosterolemia. Plant sterols. Premature coronary artery disease. Macrothrombocytopenia. ABCG5. ABCG8.

Mesh:

Substances:

Year:  2017        PMID: 29055934     DOI: 10.5604/01.3001.0010.5290

Source DB:  PubMed          Journal:  Ann Hepatol        ISSN: 1665-2681            Impact factor:   2.400


  8 in total

Review 1.  ABCG5 and ABCG8: more than a defense against xenosterols.

Authors:  Shailendra B Patel; Gregory A Graf; Ryan E Temel
Journal:  J Lipid Res       Date:  2018-05-04       Impact factor: 5.922

2.  Methodological quality and synthesis of case series and case reports.

Authors:  Mohammad Hassan Murad; Shahnaz Sultan; Samir Haffar; Fateh Bazerbachi
Journal:  BMJ Evid Based Med       Date:  2018-02-02

3.  Sitosterolemia-10 years observation in two sisters.

Authors:  Lara Veit; Gabriella Allegri Machado; Céline Bürer; Oliver Speer; Johannes Häberle
Journal:  JIMD Rep       Date:  2019-05-28

Review 4.  Acute Coronary Syndrome Developed in a 17-year-old Boy with Sitosterolemia Comorbid with Takayasu Arteritis: A Rare Case Report and Review of the Literature.

Authors:  Keita Iyama; Satoshi Ikeda; Seiji Koga; Tsuyoshi Yoshimuta; Hiroaki Kawano; Sosuke Tsuji; Koji Ando; Kayoko Matsushima; Hayato Tada; Masa-Aki Kawashiri; Atsushi Kawakami; Koji Maemura
Journal:  Intern Med       Date:  2021-10-05       Impact factor: 1.282

5.  Seventeen years of misdiagnosis in rare dyslipidaemia: a case report of sitosterolaemia in a young female.

Authors:  Tanja Charlotte Frederiksen; Martin Bødtker Mortensen; Helle Lynge Kanstrup
Journal:  Eur Heart J Case Rep       Date:  2021-05-19

Review 6.  Recent advances in understanding liver fibrosis: bridging basic science and individualized treatment concepts.

Authors:  Ralf Weiskirchen; Sabine Weiskirchen; Frank Tacke
Journal:  F1000Res       Date:  2018-06-27

Review 7.  ABCG5/G8: a structural view to pathophysiology of the hepatobiliary cholesterol secretion.

Authors:  Aiman A Zein; Rupinder Kaur; Toka O K Hussein; Gregory A Graf; Jyh-Yeuan Lee
Journal:  Biochem Soc Trans       Date:  2019-10-31       Impact factor: 4.919

Review 8.  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

  8 in total

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