| Literature DB >> 25121381 |
P M Tamhankar1, S Iyer, S Sanghavi, U Khopkar.
Abstract
Chanarin-Dorfman syndrome (CDS) is a multisystem, autosomal recessive genetic disorder characterized by congenital non-bullous ichthyosiform erythroderma with accumulation of lipid droplets in granulocytes and basal keratinocytes. An 18-month-old female child presented with typical dermatological features of CDS. She was born as a collodion baby. Liver biopsy showed micronodular cirrhosis along with macrovesicular hepatic steatosis. Sequencing of all exons and exon-intron boundaries of the ABHD5 gene showed that the patient was homozygous for a novel mutation g.24947delG (c.773 + 1delG) in intron 5. This is the first Indian child with mutation proven CDS.Entities:
Mesh:
Substances:
Year: 2014 PMID: 25121381 DOI: 10.4103/0022-3859.138826
Source DB: PubMed Journal: J Postgrad Med ISSN: 0022-3859 Impact factor: 1.476