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一名患有多发性硬化症且接受干扰素β-1a治疗并伴有单克隆丙种球蛋白病的患者出现硬化性黏液水肿。

Scleromyxedema in a patient with multiple sclerosis and monoclonal gammopathy on interferon beta-1a.

作者信息

Kumar Neeraj, Rodriguez Moses

机构信息

Department of Neurology, Mayo Medical and Graduate Schools, Rochester, MN 55905, USA.

出版信息

Mult Scler. 2004 Feb;10(1):85-6. doi: 10.1191/1352458504ms987cr.

Abstract

BACKGROUND

Animal studies have shown that some human monoclonal antibodies promote myelin repair in models of demyelinating disease. Scleromyxedema is a dermatologic disorder associated with a monoclonal gammopathy and neurologic manifestations. The reason for occurrence of cutaneous reactions in interferon treated patients is unknown.

CASE DESCRIPTION

A 37-year-old woman was started on weekly interferon beta-1a (IFN beta-1a) following a diagnosis of multiple sclerosis (MS). After having been on interferon therapy for three years, she developed skin lesions secondary to scleromyxedema. Her IFN beta-1a was discontinued and intravenous immunoglobulin therapy was started for her scleromyxedema. At a six-month follow up, her skin lesions improved and there was no recurrence of neurologic symptoms.

CONCLUSIONS

This is the first report of occurrence of scleromyxedema in a patient with MS. While this could be a chance association, it does raise the question if her neurologic manifestations could be secondary to scleromyxedema. Further research into the mechanism of IFN related cutaneous side effects is needed. Evidence regarding the remyelinating nature of human monoclonal antibodies raises interest in the potential therapeutic role these antibodies may have.

摘要

背景

动物研究表明,一些人源单克隆抗体可促进脱髓鞘疾病模型中的髓鞘修复。硬化性黏液水肿是一种与单克隆丙种球蛋白病及神经系统表现相关的皮肤病。干扰素治疗患者出现皮肤反应的原因尚不清楚。

病例描述

一名37岁女性在被诊断为多发性硬化症(MS)后开始每周注射一次干扰素β-1a(IFNβ-1a)。接受干扰素治疗三年后,她出现了继发于硬化性黏液水肿的皮肤病变。她停用了IFNβ-1a,并开始接受静脉注射免疫球蛋白治疗其硬化性黏液水肿。在六个月的随访中,她的皮肤病变有所改善,且神经症状未复发。

结论

这是首例MS患者发生硬化性黏液水肿的报告。虽然这可能是偶然关联,但确实引发了一个问题,即她的神经表现是否可能继发于硬化性黏液水肿。需要进一步研究干扰素相关皮肤副作用的机制。关于人源单克隆抗体的髓鞘再生性质的证据引发了人们对这些抗体可能具有的潜在治疗作用的兴趣。

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