Mascalchi Mario, Diciotti Stefano, Giannelli Marco, Ginestroni Andrea, Soricelli Andrea, Nicolai Emanuele, Aiello Marco, Tessa Carlo, Galli Lucia, Dotti Maria Teresa, Piacentini Silvia, Salvatore Elena, Toschi Nicola
Quantitative and Functional Neuroradiology Research Program at Meyer Children Hospital and Careggi General Hospital, Florence, Italy ; "Mario Serio" Department of Experimental and Clinical Biomedical Sciences, University of Florence, Florence, Italy.
"Mario Serio" Department of Experimental and Clinical Biomedical Sciences, University of Florence, Florence, Italy ; Department of Electrical, Electronic, and Information Engineering "Guglielmo Marconi", University of Bologna, Cesena, Italy.
PLoS One. 2014 Feb 25;9(2):e89410. doi: 10.1371/journal.pone.0089410. eCollection 2014.
Spinocerebellar ataxia type 2 (SCA2) is the second most frequent autosomal dominant inherited ataxia worldwide. We investigated the capability of magnetic resonance imaging (MRI) to track in vivo progression of brain atrophy in SCA2 by examining twice 10 SCA2 patients (mean interval 3.6 years) and 16 age- and gender-matched healthy controls (mean interval 3.3 years) on the same 1.5 T MRI scanner. We used T1-weighted images and tensor-based morphometry (TBM) to investigate volume changes and the Inherited Ataxia Clinical Rating Scale to assess the clinical deficit. With respect to controls, SCA2 patients showed significant higher atrophy rates in the midbrain, including substantia nigra, basis pontis, middle cerebellar peduncles and posterior medulla corresponding to the gracilis and cuneatus tracts and nuclei, cerebellar white matter (WM) and cortical gray matter (GM) in the inferior portions of the cerebellar hemisphers. No differences in WM or GM volume loss were observed in the supratentorial compartment. TBM findings did not correlate with modifications of the neurological deficit. In conclusion, MRI volumetry using TBM is capable of demonstrating the progression of pontocerebellar atrophy in SCA2, supporting a possible role of MRI as biomarker in future trials.
2型脊髓小脑共济失调(SCA2)是全球第二常见的常染色体显性遗传性共济失调。我们通过在同一台1.5T磁共振成像(MRI)扫描仪上对10例SCA2患者(平均间隔3.6年)和16例年龄及性别匹配的健康对照者(平均间隔3.3年)进行两次检查,研究了MRI追踪SCA2患者脑萎缩体内进展情况的能力。我们使用T1加权图像和基于张量的形态计量学(TBM)来研究体积变化,并使用遗传性共济失调临床评定量表来评估临床缺陷。与对照组相比,SCA2患者中脑的萎缩率显著更高,包括黑质、脑桥基底部、小脑中脚以及与薄束和楔束及其核团相对应的延髓后部、小脑白质(WM)和小脑半球下部的皮质灰质(GM)。幕上腔室的WM或GM体积损失未观察到差异。TBM结果与神经功能缺损的改变无关。总之,使用TBM的MRI容积测量能够显示SCA2患者脑桥小脑萎缩的进展,支持MRI在未来试验中作为生物标志物的可能作用。