[Smith-Lemli-Opitz syndrome; a special defect in cholesterol metabolism]

Ned Tijdschr Geneeskd. 1996 Jul 13;140(28):1463-6.
[Article in Dutch]

Abstract

In a male neonate dysmaturity, microcephalia, a high nasal bridge, a long philtrum, broad dental ridges, schisis of the palatum molle, retrognathia, a small penis with a chorda, a small scrotum, bilateral inguinal hernia and bilateral syndactyly of the second and third toes were observed. The presence of the Smith-Lemli-Opitz (SLO) syndrome was suspected. By gas chromatography a severely decreased plasma cholesterol level (0.27 mmol/l) was found and an increased plasma 7-dehydrocholesterol level (0.24 mmol/l). The SLO syndrome is caused by a block in the cholesterol biosynthesis due to the autosomal recessive deficiency of 7-dehydrocholesterol reductase. The patient's condition improved with use of a cholesterol-enriched diet.

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Abnormalities, Multiple
  • Cholesterol / biosynthesis
  • Cholesterol / blood*
  • Chromatography, Gas
  • Dehydrocholesterols / blood
  • Genes, Recessive
  • Humans
  • Infant, Newborn
  • Male
  • Oxidoreductases / genetics
  • Oxidoreductases Acting on CH-CH Group Donors*
  • Smith-Lemli-Opitz Syndrome / blood*

Substances

  • Dehydrocholesterols
  • Cholesterol
  • 7-dehydrocholesterol
  • Oxidoreductases
  • Oxidoreductases Acting on CH-CH Group Donors
  • 7-dehydrocholesterol reductase