Saft Carsten, Hoffmann Rainer, Strassburger-Krogias Katrin, Lücke Thomas, Meves Saskia H, Ellrichmann Gisa, Krogias Christos
Department of Neurology, Huntington Centre NRW, Ruhr-University Bochum, St. Josef-Hospital, Gudrunstr. 56, 44791, Bochum, Germany,
J Neural Transm (Vienna). 2015 Jun;122(6):825-33. doi: 10.1007/s00702-014-1335-7. Epub 2014 Dec 13.
In Huntington's disease (HD), a neurodegenerative-inherited disease, chorea as the typical kind of movement disorder is described. Beside chorea, however, all other kinds of movement disturbances, such as bradykinesia, dystonia, tremor or myoclonus can occur. Aim of the current study was to investigate alterations in the echogenicity of basal ganglia structures in different Huntington's disease phenotypes. 47 patients with manifest and genetically confirmed HD were recruited. All participants underwent a thorough neurological examination. According to a previously described method, classification into predominantly choreatic, mixed or bradykinetic-rigid motor phenotypes was performed depending on subscores of the Unified Huntington's Disease Rating Scale. In addition, findings in juvenile HD were compared to adult HD. Transcranial sonography was performed by investigators blinded to clinical classification. There were no significant differences in basal ganglia echogenicities between the three phenotypes. Size of echogenic area of substantia nigra (SN) correlated positively with CAG repeat and bradykinesia subscore, and negatively with age of onset and chorea subscore. Comparing juvenile and adult HD subtypes, SN hyperechogenicity was significantly more often detectable in the juvenile form (100 vs. 29.3 %, p = 0.002). Regarding echogenicity of caudate or lentiform nuclei, no significant differences were detected. HD patients with the juvenile variant exhibit marked hyperechogenicity of substantia nigra. No significant differences in basal ganglia echogenicities between predominantly choreatic, mixed or bradykinetic-rigid motor phenotypes were detected.
亨廷顿舞蹈症(HD)是一种神经退行性遗传病,其典型的运动障碍表现为舞蹈症。然而,除舞蹈症外,还可能出现其他各种运动障碍,如运动迟缓、肌张力障碍、震颤或肌阵挛。本研究的目的是调查不同亨廷顿舞蹈症表型中基底神经节结构回声性的改变。招募了47例临床表现明显且经基因确诊的HD患者。所有参与者都接受了全面的神经学检查。根据先前描述的方法,依据统一亨廷顿舞蹈症评定量表的子评分,将患者分为以舞蹈症为主型、混合型或运动迟缓-强直型运动表型。此外,还比较了青少年型HD和成人型HD的检查结果。由对临床分类不知情的研究人员进行经颅超声检查。三种表型之间基底神经节的回声性没有显著差异。黑质(SN)回声区的大小与CAG重复序列及运动迟缓子评分呈正相关,与发病年龄及舞蹈症子评分呈负相关。比较青少年型和成人型HD亚型,青少年型HD中SN高回声的检出率显著更高(100%对29.3%,p = 0.002)。关于尾状核或豆状核的回声性,未检测到显著差异。青少年型HD患者表现出明显的黑质高回声。以舞蹈症为主型、混合型或运动迟缓-强直型运动表型之间的基底神经节回声性没有显著差异。