Sveger T
N Engl J Med. 1976 Jun 10;294(24):1316-21. doi: 10.1056/NEJM197606102942404.
We prosepctively studied 200,000 newborns to determine the frequency and clinical characteristics of alpha1-antitrypsin deficiency. One hundred and twenty Pi Z, 48 Pi SZ, two PI Z-and one Pi S-infants were identified and followed to the age of six months. Fourteen of 120 Pi Z infants had prolonged obstructive jaundice, nine with severe clinical and laboratory evidence of liver disease. Five had only laboratory evidence of liver disease. Eight other Pi Z infants had minimal abnormalities in serum bilirubin and hepatic enzyme activity and variable hepatosplenomegaly. All 22 Pi Z infants with hepatic abnormalities, two thirds of whom were made, appeared healthy at six months of age. Ninety-eight Pi Z infants did not have clinical liver disease, but liver-function tests gave abnormal results in 44 of 84 at three months, and in 36 of 60 at six months of age. The number of small-for-gestational-age infants was greater (P less than 0.001) among those with clinical liver disease. None of the 48 Pi SZ infants had clinical liver disease, but 10 of 42 at three months and one of 22 at six months of age had abnormal liver function. The Pi Z and Pi SZ phenotypes are associated with covert or readily apparent hepatic dysfunction in the first three months of life.
我们对200,000名新生儿进行了前瞻性研究,以确定α1-抗胰蛋白酶缺乏症的发生率和临床特征。共识别出120名Pi Z型、48名Pi SZ型、2名Pi Z-型和1名Pi S型婴儿,并随访至6个月龄。120名Pi Z型婴儿中有14名出现持续性梗阻性黄疸,其中9名有严重肝病的临床和实验室证据。5名仅有肝病的实验室证据。另外8名Pi Z型婴儿血清胆红素和肝酶活性仅有轻微异常,肝脾肿大情况不一。所有22名有肝脏异常的Pi Z型婴儿,其中三分之二为男性,6个月龄时看起来健康。98名Pi Z型婴儿没有临床肝病,但84名婴儿中有44名在3个月时肝功能检查结果异常,60名婴儿中有36名在6个月时肝功能检查结果异常。临床肝病患儿中小于胎龄儿的数量更多(P<0.001)。48名Pi SZ型婴儿均无临床肝病,但42名婴儿中有10名在3个月时肝功能异常,22名婴儿中有1名在6个月时肝功能异常。Pi Z型和Pi SZ型表型与出生后前三个月隐匿或明显的肝功能障碍有关。