1Department of Diagnostic Imaging, Molecular Imaging, Interventional Radiology and Radiotherapy, University Hospital Tor Vergata , Viale Oxford 81, Rome , Italy.
Int J Neurosci. 2014 Apr;124(4):261-70. doi: 10.3109/00207454.2013.836705. Epub 2013 Sep 27.
The aim of this study was to identify potential diagnostic markers of Hereditary Spastic Paraplegia (HSP). We investigated the white matter features of spastic gait (SPG)11- and SPG4-linked HSP, using diffusion tensor imaging performed with a 3-Tesla (3T) scanner. We examined four patients with SPG11 mutations, three with SPG4 mutations, and 26 healthy controls. We obtained maps of fractional anisotropy (FA) and mean diffusivity (MD), which we analyzed through both region of interest -based approach and tract-based spatial statistics (TBSS). Compared with healthy controls, SPG11 patients presented increased MD and decreased FA in the semioval centers, frontal and peritrigonal white matter, posterior limb of the internal capsule, and throughout the corpus callosum. Similar alterations were seen in the SPG4 patients at the levels of the semioval centers, the posterior limb of the internal capsule, the left cerebral pedicle, the genu and trunk of the corpus callosum, and the peritrigonal white matter on the left. No MD or FA alterations were observed in the cerebellar white matter. In a direct comparison, white matter alterations were more pronounced and widespread in HSP-SPG11 than in HSP-SPG4 patients. Joint TBSS analysis of all three groups confirmed significant widespread alterations of FA and MD values in the supratentorial white matter. This noninvasive study documented the presence of altered diffusivity in white matter in both forms of HSP, which could represent an important diagnostic marker of HSP. The association of reduced FA and increased MD in this patient population supports the interpretation of HPG as a neurodegenerative disorder.
本研究旨在确定遗传性痉挛性截瘫(HSP)的潜在诊断标志物。我们使用 3T 磁共振扫描仪进行弥散张量成像,研究了痉挛性步态(SPG)11 型和 SPG4 型 HSP 的白质特征。我们检查了 4 名 SPG11 突变患者、3 名 SPG4 突变患者和 26 名健康对照者。我们获得了各向异性分数(FA)和平均弥散度(MD)图,并通过基于感兴趣区的方法和基于束的空间统计学(TBSS)进行了分析。与健康对照组相比,SPG11 患者在半卵圆中心、额部和周边白质、内囊后肢以及胼胝体内部均出现 MD 升高和 FA 降低。SPG4 患者在半卵圆中心、内囊后肢、左侧大脑脚、胼胝体膝部和体部以及周边白质也出现了类似的改变。小脑白质未见 MD 或 FA 改变。在直接比较中,HSP-SPG11 患者的白质改变比 HSP-SPG4 患者更明显且广泛。对三组患者的 TBSS 联合分析证实,幕上白质的 FA 和 MD 值均有明显广泛的改变。这项非侵入性研究记录了两种 HSP 形式的白质弥散度改变的存在,这可能是 HSP 的一个重要诊断标志物。在该患者人群中 FA 降低和 MD 增加的关联支持了 HPG 作为一种神经退行性疾病的解释。