Puget Sound Blood Center, Seattle, WA 98104, USA.
Br J Haematol. 2011 Aug;154(4):492-501. doi: 10.1111/j.1365-2141.2011.08612.x. Epub 2011 Jun 21.
Spherocytosis is one of the most common inherited disorders, yet presents with a wide range of clinical severity. While several genes have been found mutated in patients with spherocytosis, the molecular basis for the variability in severity of haemolytic anaemia is not entirely understood. To identify candidate proteins involved in haemolytic anaemia pathophysiology, we utilized a label-free comparative proteomic approach to detect differences in red blood cells (RBCs) from normal and β-adducin (Add2) knock-out mice. We detected seven proteins that were decreased and 48 proteins that were increased in β-adducin null RBC ghosts. Since haemolytic anaemias are characterized by reticulocytosis, we compared reticulocyte-enriched samples from phenylhydrazine-treated mice with mature RBCs from untreated mice. Among the 48 proteins increased in Add2 knockout RBCs, only 11 were also increased in reticulocytes. Of the proteins decreased in Add2 knockout RBCs, α-adducin showed the greatest intensity difference, followed by SLC9A1, the sodium-hydrogen exchanger previously termed NHE1. We verified these mass spectrometry results by immunoblot. This is the first example of SLC9A1deficiency in haemolytic anaemia and suggests new insights into the mechanisms leading to fragile RBCs.
球形红细胞增多症是最常见的遗传性疾病之一,但临床表现差异很大。虽然已经发现了几种基因突变与球形红细胞增多症有关,但溶血贫血严重程度的变化的分子基础尚未完全清楚。为了确定参与溶血性贫血病理生理学的候选蛋白,我们利用无标记比较蛋白质组学方法检测了来自正常和β-辅肌动蛋白(Add2)敲除小鼠的红细胞(RBC)之间的差异。我们检测到 7 种蛋白在β-辅肌动蛋白缺失 RBC 血影中减少,48 种蛋白增加。由于溶血性贫血的特征是网织红细胞增多,我们比较了苯肼处理小鼠的网织红细胞富集样本与未处理小鼠的成熟 RBC。在 Add2 敲除 RBC 中增加的 48 种蛋白中,只有 11 种也在网织红细胞中增加。在 Add2 敲除 RBC 中减少的蛋白中,α-辅肌动蛋白的强度差异最大,其次是 SLC9A1,即以前称为 NHE1 的钠-氢交换体。我们通过免疫印迹验证了这些质谱结果。这是 SLC9A1 缺陷导致溶血性贫血的首例实例,并为导致脆弱 RBC 的机制提供了新的见解。