Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin; Right Patient Right Drug Diagnostics, Wauwatosa, Wisconsin.
Clinical Pharmacology, Toxicology, and Therapeutic Innovation, Children's Mercy Kansas City, University of Missouri-Kansas City, Kansas City, Missouri.
J Mol Diagn. 2021 May;23(5):577-588. doi: 10.1016/j.jmoldx.2021.01.013. Epub 2021 Feb 22.
Cytochrome P450 2D6 (CYP2D6) copy number (CN) variation affects the metabolism of numerous prescribed drugs. Sequence variation within primer or probe target regions of hydrolysis probe CN assays can generate false-positive calls for CN loss. Furthermore, CYP2D6-CYP2D7 hybrids and gene conversions can cause difficult to interpret discordant CN calls. The identification of haplotypes with CN variations and structural arrangements is important to predict phenotype accurately. During clinical testing with hydrolysis probe assays targeting three CYP2D6 regions (intron 2, intron 6, and exon 9), samples with haplotypes causing inconsistent CN calls were identified. To resolve these cases, next-generation sequencing and allele-specific Sanger sequencing was performed. Sequence analysis of 16 samples, all but one from subjects of African descent, identified six novel suballeles containing single-nucleotide polymorphisms, which cause false-positive calls for CN loss in introns 2 and 6. Five samples with an exon 9 CN loss contained CYP2D6/CYP2D7 hybrids (∗13 or ∗36) and one sample was found to have a novel haplotype, CYP2D6∗141. Interestingly, CYP2D6∗141 contains a CYP2D7-derived exon 9 conversion and core single-nucleotide polymorphisms that are otherwise found in CYP2D6∗17 and ∗27. Although these variants are rare, they can cause inconsistent CN calls that typically are reported as no calls or indeterminant, and thus may deprive patients, particularly those of African descent, from taking full benefit of pharmacogenetic testing.
细胞色素 P450 2D6(CYP2D6)拷贝数(CN)变异会影响许多处方药物的代谢。水解探针 CN 测定中引物或探针靶区的序列变异可导致 CN 缺失的假阳性结果。此外,CYP2D6-CYP2D7 杂种和基因转换可导致难以解释的不一致的 CN 结果。具有 CN 变异和结构排列的单倍型的鉴定对于准确预测表型很重要。在针对 CYP2D6 的三个区域(内含子 2、内含子 6 和外显子 9)的水解探针检测的临床检测中,鉴定出了导致不一致的 CN 结果的单倍型。为了解决这些问题,进行了下一代测序和等位基因特异性 Sanger 测序。对 16 个样本(除一个来自非洲裔个体)的序列分析确定了 6 个新的亚等位基因,其中包含单核苷酸多态性,可导致 2 号和 6 号内含子 CN 缺失的假阳性结果。5 个外显子 9 CN 缺失的样本含有 CYP2D6/CYP2D7 杂种(∗13 或 ∗36),1 个样本发现了一个新的单倍型,CYP2D6∗141。有趣的是,CYP2D6∗141 包含 CYP2D7 衍生的外显子 9 转换和核心单核苷酸多态性,这些多态性通常存在于 CYP2D6∗17 和 ∗27 中。尽管这些变体很少见,但它们会导致不一致的 CN 结果,通常被报告为无结果或不确定,因此可能使患者,特别是非洲裔患者,无法充分受益于药物遗传学检测。