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威尔逊氏病——目前使用的抗铜疗法。

Wilson disease - currently used anticopper therapy.

作者信息

Członkowska Anna, Litwin Tomasz

机构信息

Second Department of Neurology, Institute of Psychiatry and Neurology, Warsaw, Poland; Department of Experimental and Clinical Pharmacology, Medical University of Warsaw, Poland.

Second Department of Neurology, Institute of Psychiatry and Neurology, Warsaw, Poland.

出版信息

Handb Clin Neurol. 2017;142:181-191. doi: 10.1016/B978-0-444-63625-6.00015-X.

DOI:10.1016/B978-0-444-63625-6.00015-X
PMID:28433101
Abstract

Wilson disease (WD) is a genetic disorder of copper metabolism that can be treated successfully with pharmacologic treatment. Two groups of drugs are currently used: chelators (e.g., d-penicillamine and trientine), which increase urinary copper excretion, and zinc salts, which inhibit copper absorption in the digestive tract. The mechanisms of action lead to a negative copper balance, stopping pathologic accumulation of copper in the tissues and clearing affected organs of copper overload. Due to a lack of prospective clinical trials, the use of drugs depends mainly on center experience and the accessibility in different countries or regions. This chapter presents the different reports and recommendations regarding WD treatment. In addition to the different expert opinions on pharmacologic agents, there are a few axioms regarding WD treatment: treatment should start immediately after diagnosis, even in clinically presymptomatic cases; the patient should be treated for life, making compliance a key factor in treatment success; and the treatment should be monitored regularly via liver and hematologic tests, neurologic examination, and copper metabolism, modifying the treatment accordingly. Other drugs proposed for WD treatment (e.g., tetrathiomolybdate) are in clinical trials and lack current recommendations. Thus, only the currently available options for WD pharmacologic treatment are discussed.

摘要

威尔逊病(WD)是一种铜代谢的遗传性疾病,可通过药物治疗成功治愈。目前使用两类药物:螯合剂(如d-青霉胺和曲恩汀),可增加尿铜排泄;锌盐,可抑制消化道对铜的吸收。其作用机制导致铜平衡为负,阻止铜在组织中的病理性蓄积,并清除铜过载的受累器官。由于缺乏前瞻性临床试验,药物的使用主要取决于各中心的经验以及在不同国家或地区的可及性。本章介绍了有关WD治疗的不同报告和建议。除了对药物的不同专家意见外,关于WD治疗还有一些公理:即使在临床无症状的情况下,诊断后也应立即开始治疗;患者应终身接受治疗,使依从性成为治疗成功的关键因素;应通过肝脏和血液学检查、神经系统检查及铜代谢定期监测治疗情况,并据此调整治疗方案。其他提议用于WD治疗的药物(如四硫钼酸盐)正在进行临床试验,目前缺乏相关推荐。因此,本文仅讨论目前WD药物治疗的可用选择。

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