Amir R, Dahle E J, Toriolo D, Zoghbi H Y
Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA.
Am J Med Genet. 2000 Jan 3;90(1):69-71.
Rett syndrome (RTT) is an X-linked dominant neurodevelopmental disorder that affects females. Exclusion mapping studies using a new family with maternal inheritance of RTT defined Xq28 as the candidate region for the RTT gene. Six candidate genes were selected for mutation analysis based on their established expression patterns and known functions in the CNS. These are: Glutamate receptor subunit 3 (GLUR3), GABA receptor subunit alpha 3 (GABRA3), GABA receptor subunit e1 (GABRE1), Vacuolar ATPase subunit 1 (VATPS1, XAP3), the human homologue of plexin 3-SEX (XAP6) and the Synaptobrevin-like protein (SYBL1). Major rearrangements involving these genes were excluded by Southern analysis. No disease-causing mutations were found, but several single-nucleotide polymorphisms (SNPs) were detected. These SNPs will be useful in future linkage analysis and whole-genome association studies for other diseases. The genomic characterization of GLUR3 and GABRA3 will allow mutational analysis of these genes as candidates for other X-linked neurological disorders mapping to Xq25-Xq26 and Xq28.
雷特综合征(RTT)是一种影响女性的X连锁显性神经发育障碍。利用一个具有RTT母系遗传的新家族进行的排除性定位研究将Xq28确定为RTT基因的候选区域。基于它们在中枢神经系统中已确定的表达模式和已知功能,选择了六个候选基因进行突变分析。它们分别是:谷氨酸受体亚基3(GLUR3)、γ-氨基丁酸受体亚基α3(GABRA3)、γ-氨基丁酸受体亚基ε1(GABRE1)、液泡ATP酶亚基1(VATPS1,XAP3)、丛状蛋白3-SEX的人类同源物(XAP6)和突触小泡蛋白样蛋白(SYBL1)。通过Southern分析排除了涉及这些基因的主要重排。未发现致病突变,但检测到了几个单核苷酸多态性(SNP)。这些SNP将在未来针对其他疾病的连锁分析和全基因组关联研究中发挥作用。GLUR3和GABRA3的基因组特征将有助于对这些基因进行突变分析,以确定它们作为其他定位到Xq25-Xq26和Xq28的X连锁神经系统疾病候选基因的可能性。