Carpi Sara, Del Grosso Ambra, De Sarlo Miriam, Colagiorgio Laura, Scaccini Luca, Tonazzini Ilaria, Parlanti Gabriele, Cecchini Marco
NEST, Istituto Nanoscienze-CNR and Scuola Normale Superiore, Piazza San Silvestro 12, 56127 Pisa, Italy.
Biomedicines. 2022 Dec 6;10(12):3146. doi: 10.3390/biomedicines10123146.
Twitcher (Twi) is a neurological Krabbe disease (KD, or globoid cell leukodystrophy) spontaneous mutant line in mice. The genome of the Twi mouse presents a single nucleotide polymorphism (SNP), leading to an enzymatically inactive galactosylceramidase (Galc) protein that causes KD. In this context, mouse Twi genotyping is an essential step in KD research. To date, the genotyping method used is labor-intensive and often has ambiguous results. Here, we evaluated a novel protocol for the genotype determination of Galc mutation status in Twi mice based on the allele-discrimination real-time polymerase chain reaction (PCR). Here, DNA is extracted from Twi mice (n = 20, pilot study; n = 120, verification study) and control group (n = 10, pilot study; n = 30 verification study) and assessed by allele-discrimination real-time PCR to detect SNP c.355G>A. Using the allele-discrimination PCR, all of the samples are identified correctly with the genotype GG (wild-type, WT), GA (heterozygote, HET), or AA (homozygote, HOM) using the first analysis and no animals are not genotyped. We demonstrated that this novel method can be used to distinguish KD timely, accurately, and without ambiguity in HOM, WT, and HET animals. This protocol represents a great opportunity to increase accuracy and speed in KD research.
颤抖小鼠(Twi)是小鼠中一种神经学上的克拉伯病(KD,或球状细胞脑白质营养不良)自发突变品系。Twi小鼠的基因组存在一个单核苷酸多态性(SNP),导致产生一种无酶活性的半乳糖神经酰胺酶(Galc)蛋白,从而引发KD。在此背景下,小鼠Twi基因分型是KD研究中的关键步骤。迄今为止,所使用的基因分型方法 labor-intensive且结果往往不明确。在此,我们评估了一种基于等位基因鉴别实时聚合酶链反应(PCR)来确定Twi小鼠中Galc突变状态基因型的新方案。在此,从Twi小鼠(n = 20,预实验;n = 120,验证实验)和对照组(n = 10,预实验;n = 30,验证实验)中提取DNA,并通过等位基因鉴别实时PCR进行评估,以检测SNP c.355G>A。使用等位基因鉴别PCR,通过首次分析即可将所有样本正确鉴定为基因型GG(野生型,WT)、GA(杂合子,HET)或AA(纯合子,HOM),且没有动物无法进行基因分型。我们证明,这种新方法可用于及时、准确且明确地区分HOM、WT和HET动物中的KD。该方案为提高KD研究的准确性和速度提供了一个绝佳机会。 (注:原文中“labor-intensive”未翻译完整,推测可能是“劳动强度大的”之类意思,因不确定准确意思,保留英文供参考)