Rachmilewitz E A, Kornberg A, Acker M
Ann N Y Acad Sci. 1982;393:336-47. doi: 10.1111/j.1749-6632.1982.tb31273.x.
Plasma vitamin E levels were found to be decreased (less than 0.5 mg) in thalassemia and in 17 out of 20 patients with Gaucher's disease, where the levels were two standard deviations below the normal mean value. In the latter, the decrease in vitamin E levels correlated with the severity of the clinical expression of the disease and correlated inversely with the degree of hepatosplenomegaly and serum tartrate-resistant acid phosphatase activity. In both diseases, there was no evidence for intestinal malabsorption of the lipid-soluble vitamin. In spite of the different etiology, pathophysiology, and clinical expression, severe vitamin E deficiency could result in both diseases by a common mechanism. In thalassemia, rapid consumption of vitamin E occurs while neutralizing oxidative damage in the pathological erythrocyte membranes and in other tissues. In Gaucher's disease, lysosomal accumulation of glucocerebroside may stimulate phagocytes into a maintained "respiratory burst" with excessive production of oxygen free radicals, resulting in increased utilization and eventual deficiency of vitamin E. Efficacy of antioxidant therapy was evaluated by administration of vitamin E with and without canthaxanthin, which has similar antioxidant properties to beta-carotene, to patients with beta-thalassemia. The results showed increased serum vitamin E levels and a decrease in the extent of erythrocyte lipid membrane peroxidation, while no significant changes occurred in hemoglobin levels and in transfusion requirements.
地中海贫血患者以及20例戈谢病患者中有17例被发现血浆维生素E水平降低(低于0.5毫克),这些患者的维生素E水平比正常均值低两个标准差。在戈谢病患者中,维生素E水平的降低与疾病临床表现的严重程度相关,与肝脾肿大程度及血清抗酒石酸酸性磷酸酶活性呈负相关。在这两种疾病中,均未发现脂溶性维生素存在肠道吸收不良的证据。尽管病因、病理生理学及临床表现不同,但严重的维生素E缺乏在这两种疾病中可能是由共同机制导致的。在地中海贫血中,在中和病理性红细胞膜及其他组织中的氧化损伤时,维生素E会快速消耗。在戈谢病中,葡糖脑苷脂在溶酶体中的蓄积可能刺激吞噬细胞持续产生“呼吸爆发”,过度产生氧自由基,导致维生素E利用增加并最终缺乏。通过对β地中海贫血患者给予维生素E以及同时给予与β胡萝卜素具有相似抗氧化特性的角黄素,评估抗氧化治疗的疗效。结果显示血清维生素E水平升高,红细胞脂质膜过氧化程度降低,而血红蛋白水平及输血需求无显著变化。