Orlacchio Antonio, Kawarai Toshitaka, Gaudiello Fabrizio, St George-Hyslop Peter H, Floris Roberto, Bernardi Giorgio
Laboratorio di Neurogenetica, Centro Europeo Di Ricerca Sul Cervello-Istituto di Ricovero e cura a Carattere Scientifico Santa Lucia, Rome, Italy.
Ann Neurol. 2005 Sep;58(3):423-9. doi: 10.1002/ana.20590.
We have updated the clinical description of a large Scottish pedigree, in which patients were affected by spastic paraplegia complicated by hearing impairment and persistent vomiting due to hiatal hernia inherited as an autosomal dominant trait. Using a genome-wide mapping approach, we identified a novel locus (SPG29) for this form of hereditary spastic paraplegia on chromosome 1p31.1-21.1 and narrowed it to 22.3cM between markers D1S2889 and D1S248. Sequencing of one candidate gene in the region (sorting nexin 7, SNX7), involved in several stages of intracellular trafficking and protein transport, showed no disease-causing mutations.
我们更新了一个大型苏格兰家系的临床描述,该家系中的患者患有痉挛性截瘫,并伴有听力障碍和因食管裂孔疝导致的持续性呕吐,呈常染色体显性遗传。通过全基因组定位方法,我们在1号染色体1p31.1 - 21.1区域确定了这种遗传性痉挛性截瘫的一个新位点(SPG29),并将其缩小至标记物D1S2889和D1S248之间的22.3厘摩。对该区域一个候选基因(分选连接蛋白7,SNX7)进行测序,该基因参与细胞内运输和蛋白质转运的多个阶段,结果显示没有致病突变。