Lecomte M C, Dhermy D, Garbarz M, Feo C, Gautero H, Bournier O, Picat C, Chaveroche I, Ester A, Galand C
Hum Genet. 1985;71(4):351-7. doi: 10.1007/BF00388462.
Five patients with hereditary elliptocytosis (HE) from two unrelated black families were studied. The patients had prominent elliptocytosis and a decreased erythrocyte resistance to heat treatment. In one infant blood smears showed elliptocytosis and poikilocytosis; his erythrocytes fagmented at a lower temperature than those of his mother and sister, both having typical mild HE. Defective dimer-dimer association was present in all patients. Limited tryptic digestion of spectrin and subsequent analysis by one- and two-dimensional electrophoresis revealed a similar and reproducible decrease in the 80,000-dalton peptide (alpha I domain) and the concomitant appearance of a 46,000-dalton peptide. All the patients had the polymorphism of the spectrin alpha II domain commonly observed in black populations. In addition, modifications relative to the alpha III domain were detected; similar variants were found in one black control subject out of 136 and are likely related to a genetic polymorphism of the alpha III domain. No differences were observed between the peptide patterns in the infant with poikilocytosis and those of his HE sister and mother.
对来自两个无亲缘关系的黑人家庭的5例遗传性椭圆形红细胞增多症(HE)患者进行了研究。这些患者有明显的椭圆形红细胞增多,且红细胞对热处理的抵抗力降低。在一名婴儿中,血液涂片显示有椭圆形红细胞增多和异形红细胞症;其红细胞在比他的母亲和姐姐更低的温度下发生裂解,他的母亲和姐姐均患有典型的轻度HE。所有患者均存在二聚体 - 二聚体结合缺陷。对血影蛋白进行有限的胰蛋白酶消化,随后通过一维和二维电泳分析,发现80,000道尔顿肽(αI结构域)出现类似且可重复的减少,并伴随出现46,000道尔顿的肽。所有患者均具有在黑人人群中常见的血影蛋白αII结构域多态性。此外,检测到相对于αIII结构域的修饰;在136名黑人对照受试者中有1名发现了类似的变异体,这可能与αIII结构域的基因多态性有关。在患有异形红细胞症的婴儿与其患有HE的姐姐和母亲之间,未观察到肽图谱存在差异。