Liu Ying-Na, Li Ru, Li Dong-Zhi
Prenatal Diagnostic Center, Guangzhou Maternal and Neonatal Hospital, Guangzhou Women and Children's Medical Center, Guangzhou Medical College, Guangzhou, Guangdong, China.
J Matern Fetal Neonatal Med. 2011 Jan;24(1):186-8. doi: 10.3109/14767058.2010.482621. Epub 2010 Jun 23.
Type 1 thanatophoric dysplasia (TD) is typically a lethal dwarfism. It is not always possible to distinguish fetuses with TD from other skeletal dysplasia in utero by ultrasonography. A definite diagnosis should be established by molecular genetic analysis to find out the abnormal mutations in the fibroblast growth factor receptor 3 (FGFR 3) gene. Among the known mutations of this gene, the C742T (R248C) mutation is the most common one associated with type 1 TD. Exon 7 of the FGFR3 gene was analyzed in 10 prenatal samples with type 1 TD, as well as in 30 control individuals for the presence of the c.742 C > T variant using melting curve analysis with a high-resolution melting instrument. The high-resolution melting curve analysis successfully genotyped this mutation in all 10 samples with type 1 TD without the need of further assays. The technique had a sensitivity and specificity of 100%. This study suggest that high-resolution melting analysis is a simple, rapid, and sensitive one tube assay for genotyping the FGFR3 gene.
1型致死性骨发育不全(TD)通常是一种致死性侏儒症。通过超声检查在子宫内并不总是能够将患有TD的胎儿与其他骨骼发育异常区分开来。应通过分子遗传学分析来明确诊断,以找出成纤维细胞生长因子受体3(FGFR 3)基因中的异常突变。在该基因的已知突变中,C742T(R248C)突变是与1型TD相关的最常见突变。使用高分辨率熔解仪通过熔解曲线分析,对10例患有1型TD的产前样本以及30例对照个体的FGFR3基因第7外显子进行了c.742 C>T变异检测。高分辨率熔解曲线分析成功地对所有10例1型TD样本中的该突变进行了基因分型,无需进一步检测。该技术的灵敏度和特异性均为100%。本研究表明,高分辨率熔解分析是一种用于FGFR3基因分型的简单、快速且灵敏的单管检测方法。