Ackland M L, Danks D M, McArdle H J
Scobie and Claire Mackinnon Trace Element Research Group, Murdoch Institute for Research into Birth Defects, Royal Children's Hospital, Parkville, Victoria, Australia.
Biol Trace Elem Res. 1989 Dec;22(3):257-63. doi: 10.1007/BF02916614.
Acrodermatitis enteropathica (AE) is a zinc deficiency disease. To date, the only defect has been demonstrated in the gut. We have investigated zinc uptake in fibroblasts established from four unrelated patients with AE using normal skin fibroblasts as controls. Zinc content of AE and control cells was similar (0.3 fmol/cell). Zinc accumulation over 24 h from a complete culture medium was similar in both normal controls and mutant cells. The fraction of zinc removed by Pronase treatment remained constant at 50 pmol/micrograms DNA, whereas the zinc remaining after Pronase treatment accumulated rapidly for 8 h, then more slowly. Analysis of binding data showed no significant difference between AE and control cells, with apparent Ka values of 4-6 X 10(6) M-1 and between 1 and 2 X 10(8) receptors/cell. Analysis of Pronase resistant data showed no difference between the control and the mutant cells with apparent Km values of 0.2-0.3 microM and Vmax values of 17-19 pmol/micrograms DNA/h. No difference in zinc efflux rates was detected. We conclude that the defect that underlies acrodermatitis enteropathica is either not expressed in fibroblasts or cannot be detected under these experimental conditions.
肠病性肢端皮炎(AE)是一种锌缺乏症。迄今为止,仅在肠道中发现了缺陷。我们使用正常皮肤成纤维细胞作为对照,研究了来自四名无亲缘关系的AE患者的成纤维细胞对锌的摄取情况。AE细胞和对照细胞的锌含量相似(0.3飞摩尔/细胞)。正常对照和突变细胞从完全培养基中24小时内的锌积累情况相似。经链霉蛋白酶处理后去除的锌部分保持在50皮摩尔/微克DNA不变,而链霉蛋白酶处理后剩余的锌在8小时内迅速积累,然后积累速度减慢。结合数据分析显示,AE细胞和对照细胞之间无显著差异,表观解离常数(Ka)值为4 - 6×10⁶ M⁻¹,受体数量在1 - 2×10⁸/细胞之间。对链霉蛋白酶抗性数据的分析显示,对照细胞和突变细胞之间无差异,表观米氏常数(Km)值为0.2 - 0.3微摩尔,最大反应速度(Vmax)值为17 - 19皮摩尔/微克DNA/小时。未检测到锌流出率的差异。我们得出结论,肠病性肢端皮炎潜在的缺陷要么在成纤维细胞中未表达,要么在这些实验条件下无法检测到。