Literature DB >> 33455044

Chanarin-Dorfman Syndrome: A comprehensive review.

Erol Cakmak1, Gokhan Bagci2.   

Abstract

The Chanarin-Dorfman syndrome (CDS) is a rare, autosomal recessively inherited genetic disease. This syndrome is associated with a decrease in the lipolysis activity in multiple tissue cells because of recessive mutations in the abhydrolase domain containing 5 (ABHD5) gene, which leads to the accumulation of lipid droplets in multiple types of cells. Major clinical symptoms in patients with CDS include ichthyosis and intracytoplasmic lipid droplets. The variability of clinical symptoms in patients with CDS depends on a large number of mutations involved. In this syndrome, liver involvement is an important cause of mortality and morbidity. This review aims to summarize the demographic characteristic, clinical symptoms, liver involvement and mutations in CDS patients in the literature to date.
© 2021 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  CGI-58; Chanarin-Dorfman syndrome; ichthyosis; non-alcoholic steatohepatitis

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Year:  2021        PMID: 33455044     DOI: 10.1111/liv.14794

Source DB:  PubMed          Journal:  Liver Int        ISSN: 1478-3223            Impact factor:   5.828


  3 in total

1.  Maternal Isodisomy of Chromosome 3 Combined with a De Novo Mutation in the ABHD5 Gene Causes Autosomal Recessive Chanarin-Dorfman Syndrome.

Authors:  Julia Kopp; Cristina Has; Alrun Hotz; Sarah C Grünert; Judith Fischer
Journal:  Genes (Basel)       Date:  2021-07-29       Impact factor: 4.096

2.  Case Report: Chanarin-Dorfman Syndrome: A Novel Homozygous Mutation in ABHD5 Gene in a Chinese Case and Genotype-Phenotype Correlation Analysis.

Authors:  Bo Liang; He Huang; Jiaxiang Zhang; Gang Chen; Xiangsheng Kong; Mengting Zhu; Peiguang Wang; Lili Tang
Journal:  Front Genet       Date:  2022-03-28       Impact factor: 4.599

3.  Chanarin-Dorfman syndrome treatment with acitretin.

Authors:  Reem AlNeyadi; Shaden Abdelhadi; Zbigniew Ruszczak
Journal:  JAAD Case Rep       Date:  2022-03-31
  3 in total

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