| Literature DB >> 3575262 |
M J Bennett, F Allison, G W Lowther, R G Gray, D I Johnston, J S Fitzsimmons, N J Manning, R J Pollitt.
Abstract
A fatal case of medium-chain acyl-coenzyme A dehydrogenase deficiency is described in a patient who presented with hypoglycaemia and a gross non-ketotic dicarboxylic aciduria. Cultured skin fibroblasts released 14CO2 from [1-14C] octanoic acid at half the normal rate. Prenatal diagnosis was undertaken in a subsequent pregnancy in which cultured amniotic fluid cells revealed a marked reduction in octanoate oxidation indicative of an affected fetus. The pregnancy was terminated and the diagnosis was confirmed by enzyme analysis of skin fibroblasts taken from the fetus. The high residual octanoate oxidation by affected fibroblasts together with the absence of any characteristic abnormality of amniotic fluid organic acids are a potential limitation to the reliability of this type of prenatal diagnosis.Entities:
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Year: 1987 PMID: 3575262 DOI: 10.1002/pd.1970070210
Source DB: PubMed Journal: Prenat Diagn ISSN: 0197-3851 Impact factor: 3.050