Department of Internal Medicine, Division of Hematology, Oncology and Cell Therapy, Rush University Medical Center, Chicago, Illinois, United States of America.
Department of Pathology, Division of Medical Laboratory Science, Rush University Medical Center, Chicago, Illinois, United States of America.
PLoS One. 2018 Apr 16;13(4):e0195497. doi: 10.1371/journal.pone.0195497. eCollection 2018.
Hereditary breast and ovarian cancer syndrome (HBOC) is most frequently caused by mutations in BRCA1 or BRCA2 (in short, BRCA) genes. The incidence of hereditary breast and ovarian cancer in relatives of BRCA mutation carriers who test negative for the familial mutation (non-carriers) may be increased. However, the data is controversial, and at this time, these individuals are recommended the same cancer surveillance as general population. One possible explanation for BRCA phenocopies (close relatives of BRCA carriers who have developed cancer consistent with HBOC but tested negative for a familial mutation) is natural chimerism where lack of detectable mutation in blood may not rule out the presence of the mutation in the other tissues. To test this hypothesis, archival tumor tissue from eleven BRCA phenocopies was investigated. DNA from the tumor tissue was analyzed using sequence-specific PCR, capillary electrophoresis, and pyrosequencing. The familial mutations were originally detected in the patients' first-degree relatives by commercial testing. The same testing detected no mutations in the blood of the patients under study. The test methods targeted only the known familial mutation in the tumor tissue. Tumor diagnoses included breast, ovarian, endometrial and primary peritoneal carcinoma. None of the familial mutations were found in the tumor samples tested. These results do not support, but do not completely exclude, the possibility of chimerism in these patients. Further studies with comprehensive sequence analysis in a larger patient group are warranted as a chimeric state would further refine the predictive value of genetic testing to include BRCA phenocopies.
遗传性乳腺癌和卵巢癌综合征(HBOC)最常由 BRCA1 或 BRCA2(简称 BRCA)基因突变引起。BRCA 基因突变携带者的家族性突变检测阴性(非携带者)的亲属中遗传性乳腺癌和卵巢癌的发病率可能会增加。然而,数据存在争议,目前这些个体被推荐与一般人群一样进行癌症监测。BRCA 表型模拟(BRCA 携带者的密切亲属已发生符合 HBOC 的癌症,但家族性突变检测为阴性)的一个可能解释是天然嵌合体,即血液中无法检测到突变并不排除其他组织存在突变。为了验证这一假说,对 11 例 BRCA 表型模拟的存档肿瘤组织进行了研究。使用序列特异性 PCR、毛细管电泳和焦磷酸测序分析肿瘤组织中的 DNA。最初通过商业检测在患者的一级亲属中检测到家族性突变。在研究中的患者的血液中,同样的检测没有发现突变。该检测方法仅针对肿瘤组织中已知的家族性突变。肿瘤诊断包括乳腺癌、卵巢癌、子宫内膜癌和原发性腹膜癌。在测试的肿瘤样本中均未发现家族性突变。这些结果不支持,但也不完全排除这些患者存在嵌合体的可能性。由于嵌合体状态将进一步完善遗传检测的预测价值,包括 BRCA 表型模拟,因此需要在更大的患者群体中进行全面序列分析的进一步研究。