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李-佛美尼综合征:诊断不再简单——低等位基因频率致病变异的解读及种系 PV、嵌合体和克隆性造血之间的差异。

Li-Fraumeni syndrome: not a straightforward diagnosis anymore-the interpretation of pathogenic variants of low allele frequency and the differences between germline PVs, mosaicism, and clonal hematopoiesis.

机构信息

Division of Hematology and Oncology, Beth Israel Deaconess Medical Center, Harvard Medical School, 330 Brookline Ave, Boston, MA, 02215, USA.

Dana-Farber Cancer Institute, 450 Brookline Ave, Boston, MA, 02215, USA.

出版信息

Breast Cancer Res. 2019 Sep 18;21(1):107. doi: 10.1186/s13058-019-1193-1.

DOI:10.1186/s13058-019-1193-1
PMID:31533767
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6749714/
Abstract

The introduction of next-generation sequencing has resulted in testing multiple genes simultaneously to identify inherited pathogenic variants (PVs) in cancer susceptibility genes. PVs with low minor allele frequencies (MAFs) (< 25-35%) are highlighted on germline genetic test reports. In this review, we focus on the challenges of interpreting PVs with low MAF in breast cancer patients undergoing germline testing and the implications for management.The clinical implications of a germline PV are substantial. For PV carriers in high-penetrance genes like BRCA1, BRCA2, and TP53, prophylactic mastectomy is often recommended and radiation therapy avoided when possible for those with Li-Fraumeni syndrome (LFS). For germline PV carriers in more moderate-risk genes such as PALB2, ATM, and CHEK2, annual breast MRI is recommended and prophylactic mastectomies considered for those with significant family histories. Detection of PVs in cancer susceptibility genes can also lead to recommendations for other prophylactic surgeries (e.g., salpingo-oophorectomy) and increased surveillance for other cancers. Therefore, recognizing when a PV is somatic rather than germline and distinguishing somatic mosaicism from clonal hematopoiesis (CH) is essential. Mutational events that occur at a post-zygotic stage are somatic and will only be present in tissues derived from the mutated cell, characterizing classic mosaicism. Clonal hematopoiesis is a form of mosaicism restricted to the hematopoietic compartment.Among the genes in multi-gene panels used for germline testing of breast cancer patients, the detection of a PV with low MAF occurs most often in TP53, though has been reported in other breast cancer susceptibility genes. Distinguishing a germline TP53 PV (LFS) from a somatic PV (TP53 mosaicism or CH) has enormous implications for breast cancer patients and their relatives.We review how to evaluate a PV with low MAF. The identification of the PV in another tissue confirms mosaicism. Older age, exposure to chemotherapy, radiation, and tobacco are known risk factors for CH, as is the absence of a LFS-related cancer in the setting of a TP53 PV with low MAF. The ability to recognize and understand the implications of somatic PVs, including somatic mosaicism and CH, enables optimal personalized care of breast cancer patients.

摘要

下一代测序技术的引入使得同时检测多个基因以鉴定癌症易感性基因中的遗传致病性变异(PVs)成为可能。具有低次要等位基因频率(MAF)(<25-35%)的 PVs 在种系遗传检测报告中被突出显示。在这篇综述中,我们重点讨论了在进行种系检测的乳腺癌患者中解释低 MAF 的 PV 所面临的挑战,以及这些挑战对管理的影响。

种系 PV 的临床意义重大。对于 BRCA1、BRCA2 和 TP53 等高外显率基因中的 PV 携带者,常建议预防性乳房切除术,并尽可能避免 Li-Fraumeni 综合征(LFS)患者的放射治疗。对于 PALB2、ATM 和 CHEK2 等中等风险基因中的种系 PV 携带者,建议每年进行乳房 MRI 检查,并考虑对具有显著家族史的患者进行预防性乳房切除术。在癌症易感性基因中检测到 PV 还可能导致对其他预防性手术(如输卵管卵巢切除术)的建议,并增加对其他癌症的监测。因此,识别 PV 是体细胞而非种系的,以及区分体细胞嵌合体和克隆性造血(CH)是至关重要的。发生在合子后阶段的突变事件是体细胞的,仅存在于源自突变细胞的组织中,这是经典嵌合体的特征。克隆性造血是一种仅限于造血区室的嵌合体形式。

在用于乳腺癌患者种系检测的多基因面板中的基因中,最常检测到具有低 MAF 的 TP53 PV,尽管在其他乳腺癌易感性基因中也有报道。区分种系 TP53 PV(LFS)和体细胞 PV(TP53 嵌合体或 CH)对乳腺癌患者及其亲属具有巨大影响。我们回顾了如何评估低 MAF 的 PV。在另一种组织中鉴定出该 PV 可确认嵌合体。已知的 CH 危险因素包括年龄较大、化疗、放疗和吸烟,以及在低 MAF 的 TP53 PV 中不存在 LFS 相关癌症。能够识别和理解体细胞 PV 的影响,包括体细胞嵌合体和 CH,能够为乳腺癌患者提供最佳的个性化护理。

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