Hereditary Cancer Program, Molecular Mechanisms and Experimental Therapy in Oncology (Oncobell) Program, Catalan Institute of Oncology, IDIBELL, Av. Gran Via 199-203, 08908, L'Hospitalet de Llobregat, Spain.
Centro de Investigación Biomédica en Red de Cáncer (CIBERONC), 28029, Madrid, Spain.
Sci Rep. 2021 Nov 25;11(1):22948. doi: 10.1038/s41598-021-02465-y.
Case-control studies have shown an association of BARD1 with hereditary breast and/or ovarian cancer (HBOC) predisposition. BARD1 alternatively spliced isoforms are abundant and some are highly expressed in different cancer types. In addition, a number of BARD1 germline pathogenic variants have been reported among HBOC patients. In previous reports, BARD1 c.1977A>G variant has been classified as pathogenic since it produces a frameshift transcript lacking exons 2 to 9. In the present study, we sought to validate the mRNA splicing results previously published and to contribute with new evidence to refine the classification of this substitution according to ACMG/AMP guidelines. The presence of the variant was screened in patients and controls. RT-PCR was performed in order to compare the transcriptional profiles of two variant carriers and ten non-carrier controls. In addition, allele-specific expression was assessed. No differences in variant frequency were detected between patients and controls. The RNA assay confirmed the presence of the shorter transcript lacking exons 2-9, but it was detected both in carriers and non-carriers. Furthermore, allelic imbalance was discarded and no significant differences in the proportion of full-length and shorter transcript were detected between carriers and controls. The shorter transcript detected corresponds to BARD1 isoform η, constituted by exons 1, 10 and 11. Our results support that this transcript is a constitutive splicing product rather than an aberrant transcript caused by BARD1 c.1977A>G variant, and for this reason this variant should be considered as likely benign following ACMG/AMP guidelines.
病例对照研究表明,BARD1 与遗传性乳腺癌和/或卵巢癌(HBOC)易感性有关。BARD1 剪接异构体丰富,在不同的癌症类型中表达水平较高。此外,在 HBOC 患者中已经报道了许多 BARD1 种系致病性变异。在之前的报道中,BARD1 c.1977A>G 变异被归类为致病性变异,因为它产生了一种缺失外显子 2 至 9 的移码转录本。在本研究中,我们试图验证先前发表的 mRNA 剪接结果,并根据 ACMG/AMP 指南提供新的证据来细化该替代的分类。在患者和对照组中筛选该变异的存在。进行 RT-PCR 以比较两个变异携带者和十个非携带者对照的转录谱。此外,评估了等位基因特异性表达。在患者和对照组之间未检测到变异频率的差异。RNA 分析证实了缺乏外显子 2-9 的较短转录本的存在,但在携带者和非携带者中均检测到。此外,排除了等位基因失衡,并且在携带者和对照组之间未检测到全长和较短转录本的比例存在显著差异。检测到的较短转录本对应于 BARD1 异构体 η,由外显子 1、10 和 11 组成。我们的结果支持该转录本是一种组成性剪接产物,而不是由 BARD1 c.1977A>G 变异引起的异常转录本,因此根据 ACMG/AMP 指南,该变异应被视为可能良性。