Bennett M J, Allison F, Lowther G W, Gray R G, Johnston D I, Fitzsimmons J S, Manning N J, Pollitt R J
Prenat Diagn. 1987 Feb;7(2):135-41. doi: 10.1002/pd.1970070210.
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.
本文描述了一例中链酰基辅酶A脱氢酶缺乏症的致命病例,该患者表现为低血糖和严重的非酮症性二羧酸尿症。培养的皮肤成纤维细胞从[1-14C]辛酸释放14CO2的速率仅为正常速率的一半。在随后的一次妊娠中进行了产前诊断,培养的羊水细胞显示辛酸氧化显著降低,表明胎儿受影响。妊娠终止,通过对取自胎儿的皮肤成纤维细胞进行酶分析证实了诊断。受影响的成纤维细胞对辛酸的高残留氧化以及羊水有机酸无任何特征性异常,是这类产前诊断可靠性的一个潜在限制因素。