Department of Chemistry, University of Illinois at Chicago, Chicago, IL 60607, USA.
Br J Haematol. 2009 Nov;147(3):392-5. doi: 10.1111/j.1365-2141.2009.07876.x. Epub 2009 Aug 31.
The functional roles of residues 21-43 and 55-59 in the alpha-spectrin N-terminal region in forming tetramers were determined by the introduction of mutations at each of these positions. We measured association affinities for tetramer formation (K(d)), which can be used to predict clinical severity, of these mutants. A total of nine residues critical for association with beta-spectrin were found. The mutations of six of these residues have already been known to cause hereditary elliptocytosis or hereditary pyropoikilocytosis. Clinical symptoms associated with three mutations of residues 23, 57 and 58 have not yet been reported. We suggest that these mutations may also introduce abnormalities to erythrocytes.
通过在这些位置引入突变,确定了 α- spectrin N 端区域残基 21-43 和 55-59 在形成四聚体中的功能作用。我们测量了这些突变体形成四聚体的亲和力(Kd),这可用于预测临床严重程度。总共发现了 9 个对与 β- spectrin 结合至关重要的残基。其中 6 个残基的突变已被证实可导致遗传性椭圆形红细胞增多症或遗传性热不稳定血红蛋白病。与残基 23、57 和 58 的 3 个突变相关的临床症状尚未报道。我们推测这些突变也可能导致红细胞异常。