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低剂量口服甲氨蝶呤后发生的播散性坏死性白质脑病。

Disseminated necrotizing leukoencephalopathy following low-dose oral methotrexate.

作者信息

Raghavendra S, Nair M D, Chemmanam T, Krishnamoorthy T, Radhakrishnan V V, Kuruvilla A

机构信息

Department of Neurology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Trivandrum, India.

出版信息

Eur J Neurol. 2007 Mar;14(3):309-14. doi: 10.1111/j.1468-1331.2006.01659.x.

DOI:10.1111/j.1468-1331.2006.01659.x
PMID:17355553
Abstract

Leukoencephalopathy is a recognized complication with intrathecal or intravenous methotrexate (MTX). We report a 59-year-old lady who developed MTX leukoencephalopathy with long-term low-dose oral MTX. She developed posterior leukoencephalopathy (PLE) that initially was reversible on discontinuation of oral MTX. Four months later, she developed disseminated necrotizing leukoencephalopathy (DNL), and was left with devastating neurological deficits. The sequential conventional magnetic resonance imaging (MRI), diffusion weighted imaging (DWI), MR perfusion (MRP) and MR spectroscopic (MRS) changes are highlighted in this report. MRP and MRS showed more wide spread abnormalities than DWI. Stereotactic biopsy from the lesion revealed demyelination with macrophagic infiltration, pericapillary lymphomononuclear aggregation, fibrinoid changes in the capillaries and neovascularization. Of the two cases of PLE with oral MTX reported in literature, one reversed clinically and radiologically with the discontinuation of MTX. To the best of our knowledge, this is the first reported case of DNL following oral MTX in the world literature.

摘要

白质脑病是鞘内或静脉注射甲氨蝶呤(MTX)的一种公认并发症。我们报告了一位59岁女性,她因长期低剂量口服MTX而发生MTX白质脑病。她出现了后部白质脑病(PLE),最初在停用口服MTX后是可逆的。四个月后,她发展为播散性坏死性白质脑病(DNL),并留下严重的神经功能缺损。本报告重点介绍了连续的传统磁共振成像(MRI)、扩散加权成像(DWI)、磁共振灌注(MRP)和磁共振波谱(MRS)变化。MRP和MRS显示的异常比DWI更广泛。病变的立体定向活检显示脱髓鞘伴巨噬细胞浸润、毛细血管周围淋巴细胞单核细胞聚集、毛细血管纤维蛋白样改变和新生血管形成。文献报道了两例口服MTX导致的PLE病例,其中一例在停用MTX后临床和影像学上均有好转。据我们所知,这是世界文献中首例口服MTX后发生DNL的报告病例。

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