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[威尔逊氏病]

[Wilsons disease].

作者信息

Mareček Z, Brůha R

机构信息

KlinMed s.r.o., Praha.

出版信息

Vnitr Lek. 2013 Jul;59(7):578-83.

Abstract

Wilsons disease is an autosomal recessive genetic disorder in which copper accumulates in tissues, especially in the liver and the brain. The genetic defect affects the P type ATPase gene (ATP7B). More than 500 mutations causing Wilsons disease have been described. The most common mutation in Central Europe concerns H1069Q. The symptoms of Wilsons disease include hepatic or neurological conditions. The hepatic condition is manifested as steatosis, acute or chronic hepatitis or cirrhosis. The neurological conditions are most often manifested after the age of 20 as motor disorders (tremor, speech and writing disorders), which may result in severe extrapyramidal syndrome with rigidity, dysarthria and muscle contractions. The dia-gnosis is based on clinical and laboratory assessments (neurological signs, liver lesions, low ceruloplasmin, increased free serum copper, high Cu volumes in urine, KayserFleischer ring). The dia-gnosis is confirmed by a high Cu level in liver tissue or genetic proof. Untreated Wilsons disease causes death of the patient. If treated properly the survival rate approximates to the survival rate of the common population. The treatment concerns either removal of copper from the body using chelating agents excreted into the urine (Penicillamine, Trientine) or limitation of copper absorption from the intestine and reducing the toxicity of copper (zinc, ammonium tetrathiomolybdate). In the Czech Republic, Penicillamine or zinc is used. A liver transplant is indicated in patients with fulminant hepatic failure or decompensated liver cirrhosis. In the family all siblings of the affected individual need to be screened in order to treat any asymptomatic subjects.

摘要

威尔逊氏病是一种常染色体隐性遗传病,铜在组织中蓄积,尤其是在肝脏和大脑。这种基因缺陷影响P型ATP酶基因(ATP7B)。已描述了500多种导致威尔逊氏病的突变。中欧最常见的突变是H1069Q。威尔逊氏病的症状包括肝脏或神经系统疾病。肝脏疾病表现为脂肪变性、急性或慢性肝炎或肝硬化。神经系统疾病最常于20岁以后出现,表现为运动障碍(震颤、言语和书写障碍),可能导致严重的锥体外系综合征,伴有僵硬、构音障碍和肌肉收缩。诊断基于临床和实验室评估(神经体征、肝脏病变、低铜蓝蛋白、血清游离铜升高、尿铜含量高、凯泽-弗莱舍尔环)。肝脏组织中高铜水平或基因检测可确诊。未经治疗的威尔逊氏病会导致患者死亡。如果治疗得当,生存率接近普通人群。治疗方法包括使用经尿液排泄的螯合剂(青霉胺、曲恩汀)从体内去除铜,或限制肠道对铜的吸收并降低铜的毒性(锌、四硫代钼酸铵)。在捷克共和国,使用青霉胺或锌。暴发性肝衰竭或失代偿性肝硬化患者需要进行肝移植。在家族中,所有患病个体的兄弟姐妹都需要接受筛查,以便治疗任何无症状的个体。

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