Literature DB >> 25121381

Chanarin-Dorfman syndrome: clinical report and novel mutation in ABHD5 gene.

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.

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Year:  2014        PMID: 25121381     DOI: 10.4103/0022-3859.138826

Source DB:  PubMed          Journal:  J Postgrad Med        ISSN: 0022-3859            Impact factor:   1.476


  3 in total

1.  Erratum: Chanarin-Dorfman syndrome: Clinical report and novel mutation in ABHD5 gene.

Authors: 
Journal:  J Postgrad Med       Date:  2020 Jan-Mar       Impact factor: 1.476

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 with Absent Jordan's Anomaly.

Authors:  Sandeep Arora; Shuvendu Roy; Divya Arora; Chetan Patil; Arun Kumar Jain
Journal:  Indian J Dermatol       Date:  2017 Sep-Oct       Impact factor: 1.494

  3 in total

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