Wilson病中与低铜血症相关的药物难治性癫痫发作

IF 2.3 Q2 CLINICAL NEUROLOGY
Sanyam Mahajan, Himanshu Chauhan, Vivek Singh, Vimal Kumar Paliwal
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引用次数: 0

摘要

威尔逊病是一种由ATP7B基因突变引起的铜代谢遗传性疾病,该基因编码一种负责将多余的铜排泄到胆汁和血浆中的蛋白质。过量的铜沉积在肝脏、大脑和角膜中。铜在大脑中的沉积导致皮层/基底神经节/脑干的神经元丢失、神经胶质瘤和空化。皮质病变增加了患者癫痫发作的可能性。威尔逊病癫痫发作的其他原因包括铜螯合药物引起的急性铜转移和吡哆醇缺乏症和肝性脑病。无铜饮食和铜螯合药物有时会引起缺铜,导致贫血、血小板减少、神经病变和髓神经病变。低铜血症很少引起癫痫发作。我们描述了一位患有Wilson病的患者,他出现了难治性局灶性癫痫发作,伴有意识受损,对多种抗癫痫药物,咪达唑仑,静脉注射甲基强的松龙(鉴于最近新发作的难治性癫痫持续状态),肠外吡哆醇和肝性脑病治疗有耐药性。补充铜后,他的癫痫发作迅速好转。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Hypocupraemia-related drug-refractory seizures in Wilson disease.

Wilson disease is an inherited disorder of copper metabolism caused by an ATP7B gene mutation, which encodes a protein responsible for excreting excess copper into bile and plasma. The excess copper deposits in the liver, brain and cornea. Copper deposition in the brain causes neuronal loss, gliosis and cavitation in the cortex/basal ganglia/brainstem. Cortical lesions increase patients' liability to seizures. Other causes of seizures in Wilson disease include acute copper shifts and pyridoxine deficiency produced by copper chelating drugs and hepatic encephalopathy. Copper-free diets and copper chelating drugs sometimes cause copper deficiency, resulting in anaemia, thrombocytopenia, neuropathy and myeloneuropathy. Hypocupraemia may rarely cause seizures. We describe a patient with Wilson disease who developed refractory focal seizures with impaired awareness that were resistant to multiple antiseizure medications, midazolam, intravenous methylprednisolone (given in view of recent new-onset refractory status epilepticus), parenteral pyridoxine and the treatment for hepatic encephalopathy. His seizures improved promptly with copper supplementation.

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来源期刊
PRACTICAL NEUROLOGY
PRACTICAL NEUROLOGY Medicine-Neurology (clinical)
CiteScore
3.70
自引率
3.60%
发文量
113
期刊介绍: The essential point of Practical Neurology is that it is practical in the sense of being useful for everyone who sees neurological patients and who wants to keep up to date, and safe, in managing them. In other words this is a journal for jobbing neurologists - which most of us are for at least part of our time - who plough through the tension headaches and funny turns week in and week out. Primary research literature potentially relevant to routine clinical practice is far too much for any neurologist to read, let alone understand, critically appraise and assimilate. Therefore, if research is to influence clinical practice appropriately and quickly it has to be digested and provided to neurologists in an informative and convenient way.
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