Glader B E, Backer K, Diamond L K
N Engl J Med. 1983 Dec 15;309(24):1486-90. doi: 10.1056/NEJM198312153092404.
Abnormalities of adenosine deaminase, a critical enzyme of the purine salvage pathway, have been reported in association with immune dysfunction, acute leukemia, and hereditary hemolytic anemia. We report data showing that erythrocyte adenosine deaminase activity is also abnormal in congenital hypoplastic anemia (the Diamond-Blackfan syndrome). Adenosine deaminase activity in erythrocytes from 12 patients (mean +/- S.D., 2.20 +/- 0.77 IU per gram of hemoglobin) was substantially greater than that observed in 50 controls (0.62 +/- 0.13 IU per gram). Enzyme activity in affected patients was also greater than that seen in cord blood or in erythrocytes from patients with hemolytic anemia, acquired aplastic anemia, Fanconi's hypoplastic anemia, acquired pure red-cell aplasia, or transient erythroblastopenia of childhood. These observations indicate that erythrocyte adenosine deaminase activity may be a unique marker for identifying congenital hypoplastic anemia.
嘌呤补救途径的关键酶——腺苷脱氨酶的异常,已被报道与免疫功能障碍、急性白血病及遗传性溶血性贫血有关。我们报告的数据显示,先天性再生障碍性贫血(戴蒙德-布莱克范综合征)患者的红细胞腺苷脱氨酶活性也异常。12例患者红细胞中的腺苷脱氨酶活性(均值±标准差,每克血红蛋白2.20±0.77国际单位)显著高于50例对照者(每克血红蛋白0.62±0.13国际单位)。患病患者的酶活性也高于脐血或溶血性贫血、获得性再生障碍性贫血、范科尼贫血、获得性纯红细胞再生障碍性贫血或儿童暂时性红细胞生成减少症患者的红细胞中的酶活性。这些观察结果表明,红细胞腺苷脱氨酶活性可能是识别先天性再生障碍性贫血的一个独特标志物。