Coleman M B, Lu Z H, Smith C M, Adams J G, Harrell A, Plonczynski M, Steinberg M H
Veterans Affairs Medical Center, Jackson, Mississippi 39216.
J Clin Invest. 1995 Feb;95(2):503-9. doi: 10.1172/JCI117691.
We studied the molecular basis of transfusion-dependent hemolytic anemia in an infant who rapidly developed the phenotype of beta thalassemia major. DNA sequence of one beta-globin gene of the proband revealed two mutations, one for the moderately unstable hemoglobin (Hb) Köln and another for a novel codon 32 cytosine-thymidine-guanine-->cytosine-adenine-guanine transversion encoding a leucine-->glutamine mutation. A hydrophilic glutamine residue at beta 32 has an uncharged polar side chain that could potentially distort the B helix and provoke further molecular instability. This new hemoglobin was called Hb Medicine Lake. Biosynthesis studies showed a deficit of beta-globin synthesis with early loss of beta-globin chains. An abnormal unstable hemoglobin, globin chain, or tryptic globin peptide was not present, demonstrating the extreme lability of this novel globin. Hb Medicine Lake mRNA was present, but an aberrantly spliced message was not. Absence of an abnormal beta-globin gene in the mother makes it likely that a de novo mutation occurred in the proband. The molecular pathogenesis of Hb Medicine Lake illustrates a mechanism whereby the phenotype of a genetic disorder, like the mild hemolytic anemia associated with a hemoglobinopathy, can be modulated by a coincident mutation in the same gene.
我们研究了一名迅速发展为重型β地中海贫血表型的婴儿输血依赖型溶血性贫血的分子基础。先证者的一个β珠蛋白基因的DNA序列显示有两个突变,一个是中度不稳定血红蛋白(Hb)科隆型突变,另一个是新的密码子32胞嘧啶-胸腺嘧啶-鸟嘌呤→胞嘧啶-腺嘌呤-鸟嘌呤颠换,编码亮氨酸→谷氨酰胺突变。β32位的亲水性谷氨酰胺残基具有不带电荷的极性侧链,可能会扭曲B螺旋并引发进一步的分子不稳定。这种新的血红蛋白被称为Hb Medicine Lake。生物合成研究显示β珠蛋白合成不足,β珠蛋白链早期丢失。未发现异常不稳定的血红蛋白、珠蛋白链或胰蛋白酶消化的珠蛋白肽,表明这种新珠蛋白具有极高的不稳定性。存在Hb Medicine Lake mRNA,但未发现异常剪接的信息。母亲不存在异常的β珠蛋白基因,这使得先证者很可能发生了新发突变。Hb Medicine Lake的分子发病机制说明了一种机制,即像与血红蛋白病相关的轻度溶血性贫血这样的遗传性疾病的表型,可以通过同一基因中的巧合突变来调节。