Chu Andrew S, Chopra Kapil B, Perlmutter David H
Pediatrics.
Children's Hospital of Pittsburgh, University of Pittsburgh Medical Center, Pittsburgh, PA.
Liver Transpl. 2016 Jul;22(7):886-94. doi: 10.1002/lt.24434.
The classical form of alpha-1-antitrypsin deficiency (A1ATD) is known to cause liver disease in children and adults, but there is relatively little information about the risk of severe, progressive liver disease and the need for liver transplantation. To better understand how newly evolving pharmacological, genetic, and cellular therapies may be targeted according to risk for progressive liver disease, we sought to determine the age distribution of A1ATD as a cause of severe liver disease, as defined by the need for liver transplantation. Using 3 US liver transplantation databases for the period 1991-2012, we found 77.2% of 1677 liver transplants with a reported diagnosis of A1ATD were adults. The peak age range was 50-64 years. Using 2 of the databases which included specific A1AT phenotypes, we found that many of these adults who undergo liver transplantation with A1ATD as the diagnosis are heterozygotes and have other potential causes of liver disease, most notably obesity and ethanol abuse. However, even when these cases are excluded and only ZZ and SZ phenotypes are considered, severe liver disease requiring transplantation is more than 2.5 times as likely in adults. The analysis also showed a markedly increased risk for males. In the pediatric group, almost all of the transplants are done in children less than 5 years of age. In conclusion, A1ATD causes progressive liver disease most commonly in adults with males in the highest risk category. In the pediatric group, children less than 5 years of age are highest in risk. These results suggest that A1ATD most commonly causes liver disease by mechanisms similar to age-dependent degenerative diseases and more rarely in children by powerful modifiers. Liver Transplantation 22 886-894 2016 AASLD.
已知α-1抗胰蛋白酶缺乏症(A1ATD)的经典形式会在儿童和成人中引发肝脏疾病,但关于严重、进行性肝脏疾病的风险以及肝移植需求的信息相对较少。为了更好地了解新出现的药物、基因和细胞疗法如何根据进行性肝脏疾病的风险进行靶向治疗,我们试图确定作为严重肝脏疾病病因的A1ATD的年龄分布,严重肝脏疾病定义为需要进行肝移植。利用美国3个1991 - 2012年期间的肝移植数据库,我们发现1677例报告诊断为A1ATD的肝移植受者中,77.2%为成年人。年龄峰值范围为50 - 64岁。利用其中2个包含特定A1AT表型的数据库,我们发现许多以A1ATD为诊断进行肝移植的成年人是杂合子,并且有其他潜在的肝脏疾病病因,最显著的是肥胖和乙醇滥用。然而,即使排除这些病例,仅考虑ZZ和SZ表型,成年人发生需要移植的严重肝脏疾病的可能性仍超过2.5倍。分析还显示男性的风险显著增加。在儿科组中,几乎所有移植手术都在5岁以下儿童中进行。总之,A1ATD最常导致成年人发生进行性肝脏疾病,男性处于最高风险类别。在儿科组中,5岁以下儿童风险最高。这些结果表明,A1ATD最常通过类似于年龄依赖性退行性疾病的机制导致肝脏疾病,而在儿童中则较少由强大的修饰因子导致。《肝脏移植》2016年第22卷886 - 894页,美国肝脏研究协会。