Splendore Alessandra, Fanganiello Roberto D, Masotti Cibele, Morganti Lucas S C, Passos-Bueno M Rita
Instituto Nacional de Câncer, Divisão de Genética, Rio de Janeiro, Brazil.
Hum Mutat. 2005 May;25(5):429-34. doi: 10.1002/humu.20159.
Recently, a novel exon was described in TCOF1 that, although alternatively spliced, is included in the major protein isoform. In addition, most published mutations in this gene do not conform to current mutation nomenclature guidelines. Given these observations, we developed an online database of TCOF1 mutations in which all the reported mutations are renamed according to standard recommendations and in reference to the genomic and novel cDNA reference sequences (www.genoma.ib.usp.br/TCOF1_database). We also report in this work: 1) results of the first screening for large deletions in TCOF1 by Southern blot in patients without mutation detected by direct sequencing; 2) the identification of the first pathogenic mutation in the newly described exon 6A; and 3) statistical analysis of pathogenic mutations and polymorphism distribution throughout the gene.
最近,在TCOF1中发现了一个新的外显子,尽管它是可变剪接的,但包含在主要蛋白质异构体中。此外,该基因中大多数已发表的突变不符合当前的突变命名指南。基于这些观察结果,我们开发了一个TCOF1突变在线数据库,其中所有报告的突变均根据标准建议并参考基因组和新的cDNA参考序列进行重新命名(www.genoma.ib.usp.br/TCOF1_database)。我们还在这项工作中报告了:1) 通过Southern印迹法对直接测序未检测到突变的患者进行TCOF1大片段缺失首次筛查的结果;2) 在新描述的外显子6A中鉴定出第一个致病突变;3) 对整个基因中致病突变和多态性分布的统计分析。