Vulinovic Franca, Schaake Susen, Domingo Aloysius, Kumar Kishore Raj, Defazio Giovanni, Mir Pablo, Simonyan Kristina, Ozelius Laurie J, Brüggemann Norbert, Chung Sun Ju, Rakovic Aleksandar, Lohmann Katja, Klein Christine
Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
Institute of Neurogenetics, University of Lübeck, Lübeck, Germany; Graduate School Lübeck, Lübeck, Germany.
Parkinsonism Relat Disord. 2017 Aug;41:118-120. doi: 10.1016/j.parkreldis.2017.06.001. Epub 2017 Jun 10.
Mutations in TUBB4A have been identified to cause a wide phenotypic spectrum ranging from hereditary generalized dystonia with whispering dysphonia (DYT4) to the leukodystrophy hypomyelination syndrome with atrophy of the basal ganglia and cerebellum (H-ABC). To test for the contribution of TUBB4A mutations in different ethnicities (Spanish, Italian, Korean, Japanese), we screened 492 isolated dystonia cases for mutations in this gene and for the first time determined TUBB4A copy number variations in 336 dystonia patients. A potentially pathogenic rare 3bp-in-frame deletion was found in a patient with cervical dystonia but no copy number variations were detected in this study, suggesting that TUBB4A mutations exceedingly rarely contribute to the etiology of isolated dystonia.
已确定TUBB4A突变可导致广泛的表型谱,范围从伴有轻声性发音障碍的遗传性全身性肌张力障碍(DYT4)到伴有基底神经节和小脑萎缩的脑白质营养不良综合征(H-ABC)。为了检测TUBB4A突变在不同种族(西班牙、意大利、韩国、日本)中的作用,我们对492例孤立性肌张力障碍病例进行了该基因突变筛查,并首次测定了336例肌张力障碍患者的TUBB4A拷贝数变异。在一名颈部肌张力障碍患者中发现了一个潜在致病性的罕见3bp框内缺失,但本研究未检测到拷贝数变异,这表明TUBB4A突变极少导致孤立性肌张力障碍的病因。