Zhou H, Fischer H-P
Pathologisches Institut der Universität Bonn.
Pathologe. 2008 Feb;29(1):73-83. doi: 10.1007/s00292-007-0954-2.
Primary hemochromatosis, alpha-1-antitrypsin (AAT) deficiency, and Wilson's disease are the most common hereditary causes of unclear hepatopathy. Classical primary hemochromatosis (type I) on the basis of a homozygous mutation of the HFE gene, usually presents in adults with increasing hepatocellular siderosis and chronic progressive necroinflammatory liver disease. Homozygous AAT deficiency type PiZZ becomes manifest in newborns as a giant cell hepatitis or findings similar to bile duct atresia, in adults as chronic hepatitis or "cryptogenic cirrhosis". The heterozygous PiZ mutation can lead to PAS-positive hepatocellular AAT deposits increasing over the life time. Immunohistochemical detection of AAT deposits by specific PiZ antibodies is a highly sensitive and specific supplementary method. Molecular analysis of AAT and HFE genes in paraffin-embedded tissue or blood can confirm the diagnosis and allows the zygosity status to be defined. Wilson's disease has to be considered in children and young adults with unexplained histologic findings of chronic hepatitis or steatohepatitis. Rhodanin staining is the most effective histochemical method to detect free copper deposits, but negative staining results do not exclude Wilson's disease. In cases suspected of Wilson's disease further clinical exploration must be initiated. The diagnosis is based on a combination of clinical and biochemical findings, which can be supplemented by mutation analysis of the ATP7B gene.
原发性血色素沉着症、α-1抗胰蛋白酶(AAT)缺乏症和威尔逊氏病是不明原因肝病最常见的遗传病因。基于HFE基因纯合突变的经典原发性血色素沉着症(I型),通常在成人中表现为肝细胞铁沉积增加和慢性进行性坏死性炎症性肝病。纯合子AAT缺乏症PiZZ型在新生儿中表现为巨细胞性肝炎或类似胆管闭锁的表现,在成人中表现为慢性肝炎或“隐源性肝硬化”。杂合子PiZ突变可导致PAS阳性的肝细胞AAT沉积物在一生中逐渐增加。用特异性PiZ抗体免疫组化检测AAT沉积物是一种高度敏感和特异的辅助方法。对石蜡包埋组织或血液中的AAT和HFE基因进行分子分析可确诊并确定其纯合状态。对于有不明原因的慢性肝炎或脂肪性肝炎组织学表现的儿童和年轻人,必须考虑威尔逊氏病。罗丹宁染色是检测游离铜沉积最有效的组织化学方法,但染色结果为阴性并不能排除威尔逊氏病。对于疑似威尔逊氏病的病例,必须展开进一步的临床检查。诊断基于临床和生化检查结果的综合判断,ATP7B基因突变分析可作为补充。