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选择性基因检测后的家族沟通与级联检测

Familial communication and cascade testing following elective genomic testing.

作者信息

Adelson Sophia M, Blout Zawatsky Carrie L, Hickingbotham Madison R, Bell Megan E, Platt Dylan M, Leonhard Jennifer R, Zoltick Emilie S, Hajek Catherine A, Green Robert C, Christensen Kurt D

机构信息

Brigham and Women's Hospital, Boston, Massachusetts, USA.

Ariadne Labs, Boston, Massachusetts, USA.

出版信息

J Genet Couns. 2025 Feb;34(1):e1907. doi: 10.1002/jgc4.1907. Epub 2024 May 16.

Abstract

Familial communication of results and cascade genetic testing (CGT) can extend the benefits of genetic screening beyond the patient to their at-risk relatives. While an increasing number of health systems are offering genetic screening as an elective clinical service, data are limited about how often results are shared and how often results lead to CGT. From 2018 to 2022, the Sanford Health system offered the Sanford Chip, an elective genomic test that included screening for medically actionable predispositions for disease recommended by the American College of Medical Genetics and Genomics for secondary findings disclosure, to its adult primary care patients. We analyzed patient-reported data about familial sharing of results and CGT among patients who received Sanford Chip results at least 1 year previously. Among the patients identified with medically actionable predispositions, 94.6% (53/56) reported disclosing their result to at least one family member, compared with 46.7% (423/906) of patients with uninformative findings (p < 0.001). Of the patients with actionable predispositions, 52.2% (12/23) with a monogenic disease risk and 12.1% (4/33) with a carrier status reported that their relatives underwent CGT. Results suggest that while the identification of monogenic risk during elective genomic testing motivates CGT in many at-risk relatives, there remain untested at-risk relatives who may benefit from future CGT. Findings identify an area that may benefit from increased genetic counseling and the development of tools and resources to encourage CGT for family members.

摘要

结果的家族性沟通和级联基因检测(CGT)可以将基因筛查的益处从患者扩展到其有风险的亲属。虽然越来越多的医疗系统将基因筛查作为一项选择性临床服务提供,但关于结果共享的频率以及结果导致CGT的频率的数据有限。2018年至2022年期间,桑福德医疗系统为其成年初级保健患者提供了桑福德芯片检测,这是一项选择性基因组检测,包括筛查美国医学遗传学与基因组学学会推荐的、用于次要结果披露的、具有医学可干预疾病易感性的基因。我们分析了至少在1年前收到桑福德芯片检测结果的患者报告的关于结果的家族性共享和CGT的数据。在被确定具有医学可干预易感性的患者中,94.6%(53/56)报告将其结果告知了至少一名家庭成员,而结果无意义的患者中这一比例为46.7%(423/906)(p<0.001)。在具有可干预易感性的患者中,52.2%(12/23)有单基因疾病风险且12.1%(4/33)为携带者状态的患者报告其亲属接受了CGT。结果表明,虽然在选择性基因组检测中识别出单基因风险会促使许多有风险的亲属接受CGT,但仍有未接受检测的有风险亲属可能会从未来的CGT中受益。研究结果确定了一个可能受益于加强遗传咨询以及开发鼓励家庭成员接受CGT的工具和资源的领域。

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