Dalloz M-A, Debs R, Bensa C, Alamowitch S
Service de Neurologie, Hôpital Tenon, 4 Rue de la Chine, 75020 Paris, France.
Rev Neurol (Paris). 2010 Oct;166(10):844-8. doi: 10.1016/j.neurol.2010.01.015. Epub 2010 Mar 16.
Pseudoxanthoma elasticum (PXE) is an inherited connective tissue disease characterized by skin, eye, cardiovascular, and, less often, cerebrovascular manifestations. We report the case of a 32-year-old woman who presented with fortuitously discovered cerebral white matter lesions. The pattern of the lesions was compatible with vascular leucopathy. Neurological examination, CSF and biological assessment were normal. Physical examination demonstrated cutaneous lesions characterized by yellowish papules in the neck and axilla with calcification of elastic fibres showed on the skin biopsy and retinal angioid streaks, which made PXE a plausible diagnosis. Sequencing of the ABCC6 gene confirmed PXE. Thus, PXE must be considered when confronted with cerebral microangiopathy of undetermined origin.
弹性假黄瘤(PXE)是一种遗传性结缔组织疾病,其特征为皮肤、眼部、心血管系统受累,较少累及脑血管系统。我们报告了一例32岁女性病例,该患者偶然发现脑白质病变。病变模式与血管性白质脑病相符。神经学检查、脑脊液及生物学评估均正常。体格检查发现皮肤病变,表现为颈部和腋窝出现淡黄色丘疹,皮肤活检显示弹性纤维钙化,并有视网膜血管样条纹,这些表现使弹性假黄瘤成为一个合理的诊断。ABCC6基因测序证实了弹性假黄瘤的诊断。因此,当面对病因不明的脑微血管病变时,必须考虑弹性假黄瘤。