Hubbard R C, Sellers S, Czerski D, Stephens L, Crystal R G
Pulmonary Branch, National Heart, Lung, and Blood Institute, Bethesda, MD 20892.
JAMA. 1988 Sep 2;260(9):1259-64.
The hereditary disorder alpha 1-antitrypsin (alpha 1AT) deficiency results in the development of emphysema due to a diminished anti-neutrophil elastase screen of the lower respiratory tract. Specific therapy for this disorder is available in the form of weekly intravenous infusions of human plasma alpha 1AT, which effectively reconstitute the anti-elastase screen of the lung in these individuals. In an attempt to reduce the frequency of therapy we evaluated the ability of monthly infusions of alpha 1AT to provide equivalent lower respiratory tract protection against neutrophil elastase. Intravenous infusion of 250 mg/kg of alpha 1AT at 28-day intervals to nine individuals with alpha 1AT deficiency and emphysema was carried out for 12 months. Serum alpha 1AT levels exceeded the protective threshold for an average of 25 days after each dose of alpha 1AT was administered. Furthermore, the postinfusion level of alpha 1AT in the nadir lung epithelial lining fluid was fivefold greater than the preinfusion level, and the anti-neutrophil elastase capacity of the nadir epithelial lining fluid also was elevated significantly, nearly threefold above the preinfusion level. These results indicate that monthly administration of human alpha 1AT is fully capable of adequately augmenting serum and lung alpha 1AT levels and anti-elastase capacity and is therefore a rational alternative to weekly therapy.
遗传性疾病α1-抗胰蛋白酶(α1AT)缺乏症会导致肺气肿,原因是下呼吸道的抗中性粒细胞弹性蛋白酶屏障减弱。针对这种疾病的特异性治疗方法是每周静脉输注人血浆α1AT,这能有效重建这些个体肺部的抗弹性蛋白酶屏障。为了减少治疗频率,我们评估了每月输注α1AT为下呼吸道提供等效抗中性粒细胞弹性蛋白酶保护的能力。对9名患有α1AT缺乏症和肺气肿的个体,每隔28天静脉输注250mg/kg的α1AT,持续12个月。每次输注α1AT后,血清α1AT水平平均有25天超过保护阈值。此外,最低点时肺上皮衬液中α1AT的输注后水平比输注前水平高五倍,最低点时上皮衬液的抗中性粒细胞弹性蛋白酶能力也显著升高,比输注前水平高出近三倍。这些结果表明,每月给予人α1AT完全能够充分提高血清和肺α1AT水平以及抗弹性蛋白酶能力,因此是每周治疗的合理替代方案。