Benson M D, Turpin J C, Lucotte G, Zeldenrust S, LeChevalier B, Benson M D
Indiana University School of Medicine, Department of Medicine, Indianapolis.
J Med Genet. 1993 Feb;30(2):120-2. doi: 10.1136/jmg.30.2.120.
A transthyretin (TTR) mutation is described in a 44 year old French woman from Caen who presented at the age of 40 with neuropathy in all four extremities, diarrhoea, and orthostatic hypotension. Her father died with a similar syndrome including vitreous opacities. A nerve biopsy from the proband showed amyloid deposits which stained with anti-transthyretin. Direct genomic DNA sequencing of TTR exon 3 showed both thymine and cytosine in the position corresponding to the second base of codon 71. This codes for a variant alanine (GCG) as well as the normal valine (GTG), indicating that the proband is heterozygous for the substitution. Since this substitution does not result in the creation or abolition of a restriction endonuclease recognition site, a new technique (PCR-IMRA) was used to create an RFLP. Using a 24 bp nucleotide mutagenesis primer in the PCR reaction, a new NspBII site is created on amplification of the variant allele. With this method a 170 bp TTR exon 3 PCR product was generated for both the normal and the variant allele. On digestion of the PCR product with NspBII, DNA from a heterozygous subject showed both the 170 bp undigested product from the normal allele and a 146 bp digestion product from the variant allele. By PCR-IMRA, two of five children of the proband were positive for the variant allele. This non-radioactive technique gives a rapid method for testing subjects at risk for this mutation.
在一名来自卡昂的44岁法国女性中发现了转甲状腺素蛋白(TTR)突变。该女性40岁时出现四肢神经病变、腹泻和体位性低血压。她的父亲死于类似综合征,包括玻璃体混浊。先证者的神经活检显示淀粉样沉积物,用抗转甲状腺素蛋白染色。对TTR外显子3进行直接基因组DNA测序,发现在对应于密码子71第二个碱基的位置既有胸腺嘧啶又有胞嘧啶。这编码了一个变异的丙氨酸(GCG)以及正常的缬氨酸(GTG),表明先证者在该替换位点是杂合子。由于这种替换不会导致限制性内切酶识别位点的产生或消除,因此使用了一种新技术(PCR-IMRA)来创建一个限制性片段长度多态性(RFLP)。在PCR反应中使用一个24 bp的核苷酸诱变引物,在变异等位基因扩增时创建了一个新的NspBII位点。用这种方法,正常和变异等位基因都产生了一个170 bp的TTR外显子3 PCR产物。用NspBII消化PCR产物后,杂合子个体的DNA显示出正常等位基因的170 bp未消化产物和变异等位基因的146 bp消化产物。通过PCR-IMRA,先证者的五个孩子中有两个变异等位基因呈阳性。这种非放射性技术为检测有该突变风险的个体提供了一种快速方法。