Literature DB >> 17314446

Dorfman-Chanarin syndrome.

Vijay Gandhi1, Puneet Aggarwal, Jyoti Dhawan, Usha Rani Singh, S N Bhattacharya.   

Abstract

A four-year-old girl was brought to the dermatology outpatient department with scaling all over the body since birth. She had history of episodic vomiting and abdominal distension. A dermatological diagnosis of lamellar ichthyosis was made. Abdominal examination revealed a nontender hepatomegaly, fatty liver on ultrasonography and deranged liver function tests. Peripheral blood smear showed lipid vacuoles in the granulocytes consistent with Jordans' anomaly. Similar lipid vacuoles were seen in the basal layer in skin biopsy. An inflammatory infiltrate, moderate fibrosis in the portal tract and diffuse severe fatty change in hepatocytes were seen in liver biopsy. The patient was diagnosed as a case of Dorfman-Chanarin syndrome.

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Year:  2007        PMID: 17314446     DOI: 10.4103/0378-6323.30650

Source DB:  PubMed          Journal:  Indian J Dermatol Venereol Leprol        ISSN: 0378-6323            Impact factor:   2.545


  5 in total

1.  Dorfman-chanarin syndrome: a case report.

Authors:  Seema T Methre; Ravibhushan R Godbole; Priyanka S Nayar; Rumma V Manchanda
Journal:  Indian J Hematol Blood Transfus       Date:  2011-07-06       Impact factor: 0.900

2.  A rare cause of Fatty liver and elevated aminotransferase levels: chanarin-dorfman syndrome: a case report.

Authors:  Ozdal Ersoy; Canan Alkım; Mehmet Derya Onuk; Hüseyin Demirsoy; Dilek Argon
Journal:  Int J Hepatol       Date:  2011-01-20

Review 3.  Neutral Lipid Storage Diseases as Cellular Model to Study Lipid Droplet Function.

Authors:  Sara Missaglia; Rosalind A Coleman; Alvaro Mordente; Daniela Tavian
Journal:  Cells       Date:  2019-02-21       Impact factor: 6.600

4.  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

5.  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

  5 in total

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