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Cholesteryl ester storage disease in a young child presenting as isolated hepatomegaly treated with simvastatin.

Authors :
Dalgiç B
Sari S
Gündüz M
Ezgü F
Tümer L
Hasanoğlu A
Akyol G
Source :
The Turkish journal of pediatrics [Turk J Pediatr] 2006 Apr-Jun; Vol. 48 (2), pp. 148-51.
Publication Year :
2006

Abstract

Cholesteryl ester storage disease (CESD) is an autosomal recessive disorder resulting from lysosomal acid lipase deficiency and is usually characterized by hepatomegaly and hyperlipidemia. This paper reports a two-year-old boy who had hepatosplenomegaly, hyperlipidemia and hypertransaminasemia determined incidentally. The liver biopsy sample was orange-yellow in appearance. Microscopically, microvesicular steatosis and birefringent crystals were seen in liver biopsy. The diagnosis of CESD was confirmed by the reduced human acid lipase activity in peripheral leukocytes. Simvastatin therapy was given and tolerated without side effects. Our patient is the youngest reported case in the literature treated with 3-hydroxy 3-methyl glutaryl (HMG) CoA reductase inhibitor.

Details

Language :
English
ISSN :
0041-4301
Volume :
48
Issue :
2
Database :
MEDLINE
Journal :
The Turkish journal of pediatrics
Publication Type :
Academic Journal
Accession number :
16848116