Abbruzzese G, Pigullo S, Di Maria E, Martinelli P, Barone P, Marchese R, Scaglione C, Assini A, Lucetti C, Berardelli A, Calzetti S, Bellone E, Ajmar F, Mandich P
The Italian Study Group on Essential Tremor, University of Genoa, Italy.
Neurol Sci. 2001 Feb;22(1):39-40. doi: 10.1007/s100720170036.
Essential tremor (ET) is one of the most common movement disorders. The pathogenesis is as yet unknown, although a genetic cause has long been recognised. Clinical and molecular evidence suggested that the ET gene contains a CAG expanded region. We examined a cohort of 240 Italian ET patients, classified as familial (193 cases) and sporadic (47 cases). The clinical manifestations of ET patients confirmed that the disorder is characterised by a large phenotypic variability. Repeat expansion detection (RED) approach did not demonstrate large CAG expansions. Six families were genotyped with 12 microsatellites markers of 2p and 3q regions and analysed according to parametrical methods. Lod scores values obtained in these families excluded the association of ET with 2p and 3q loci. Our findings confirm the presence of genetic heterogeneity and suggest that at least a third locus is involved in the pathogenesis of familial essential tremor.
特发性震颤(ET)是最常见的运动障碍之一。尽管长期以来人们已经认识到其存在遗传病因,但其发病机制尚不清楚。临床和分子证据表明,ET基因包含一个CAG扩展区域。我们对一组240名意大利ET患者进行了研究,这些患者分为家族性(193例)和散发性(47例)。ET患者的临床表现证实,该疾病具有很大的表型变异性。重复序列扩展检测(RED)方法未显示出大的CAG扩展。对6个家族进行了2p和3q区域的12个微卫星标记的基因分型,并根据参数方法进行了分析。在这些家族中获得的连锁分数值排除了ET与2p和3q位点的关联。我们的研究结果证实了遗传异质性的存在,并表明至少有第三个位点参与了家族性特发性震颤的发病机制。