Ouwerkerk R, van Echteld C J, Staal G E, Rijksen G
Department of Haematology, University Hospital, Utrecht, The Netherlands.
Biochim Biophys Acta. 1989 Mar 6;1010(3):294-303. doi: 10.1016/0167-4889(89)90052-9.
The erythrocyte metabolism of two patients with nonspherocytic hemolytic anemia caused by a hexokinase deficiency, and a pyruvate kinase deficiency, respectively, were studied with NMR. The complexing of ATP and 2,3-diphosphoglycerate (2,3-DPG) with Mg2+ and hemoglobin (Hb) was determined using 31P-NMR on oxygenated and deoxygenated cells to investigate the influences of these enzyme defects on intracellular magnesium distribution and on Hb oxygen dissociation. In the pyruvate kinase-deficient red blood cells, the 2,3-DPG concentration was almost twice the normal value and the ATP concentration was near the lower limit of the normal range. In the hexokinase-deficient red cell population, the predominance of young cells masked the deficiency. Therefore, reticulocyte control cells were included in this study. In the oxygenated pyruvate kinase-deficient cells, the fraction of ATP that is complexed to magnesium as well as the free Mg2+ concentration were normal, despite the abnormal concentration of 2,3-DPG. In the deoxygenated cells the free Mg2+ concentration was lower than in normal cells. The fraction of Hb complexed with 2,3-DPG was higher than normal in both oxygenated and deoxygenated pyruvate kinase-deficient cells, in accordance with the high p50 of the oxygen-hemoglobin dissociation curve. In hexokinase-deficient cells, two major abnormalities are found: when the cells were deoxygenated, the concentration of ATP and 2,3-DPG fell. This was not observed for any other sample and could, therefore, be a consequence of the hexokinase deficiency. Despite almost normal levels of magnesium-binding metabolites, the free Mg2+ concentration in oxygenated and deoxygenated cels is much lower than in normal cells. This could be a cell-age-related phenomenon, since lower free Mg2+ concentrations were also found in reticulocyte control cells.
分别对两名因己糖激酶缺乏和丙酮酸激酶缺乏导致的非球形红细胞溶血性贫血患者的红细胞代谢进行了核磁共振研究。利用31P-NMR测定了氧合和脱氧细胞中ATP和2,3-二磷酸甘油酸(2,3-DPG)与Mg2+和血红蛋白(Hb)的络合情况,以研究这些酶缺陷对细胞内镁分布和Hb氧解离的影响。在丙酮酸激酶缺乏的红细胞中,2,3-DPG浓度几乎是正常值的两倍,而ATP浓度接近正常范围的下限。在己糖激酶缺乏的红细胞群体中,年轻细胞占优势掩盖了缺陷。因此,本研究纳入了网织红细胞对照细胞。在氧合的丙酮酸激酶缺乏细胞中,尽管2,3-DPG浓度异常,但与镁络合的ATP部分以及游离Mg2+浓度正常。在脱氧细胞中,游离Mg2+浓度低于正常细胞。在氧合和脱氧的丙酮酸激酶缺乏细胞中,与2,3-DPG络合的Hb部分均高于正常水平,这与氧合血红蛋白解离曲线的高p50一致。在己糖激酶缺乏的细胞中,发现了两个主要异常:当细胞脱氧时,ATP和2,3-DPG浓度下降。其他任何样本均未观察到这种情况,因此这可能是己糖激酶缺乏的结果。尽管镁结合代谢物水平几乎正常,但氧合和脱氧细胞中的游离Mg2+浓度远低于正常细胞。这可能是一种与细胞年龄相关的现象,因为在网织红细胞对照细胞中也发现了较低的游离Mg2+浓度。