FHU TRANSLAD, Centre Hospitalier Universitaire Dijon-Bourgogne et Université de Bourgogne-Franche Comté, Dijon, France.
Inserm UMR 1231 GAD « Génétique des Anomalies du Développement », Université de Bourgogne, Dijon, France.
Eur J Hum Genet. 2019 Aug;27(8):1197-1214. doi: 10.1038/s41431-019-0384-7. Epub 2019 Apr 24.
With exome/genome sequencing (ES/GS) integrated into the practice of medicine, there is some potential for reporting incidental/secondary findings (IFs/SFs). The issue of IFs/SFs has been studied extensively over the last 4 years. In order to evaluate their implications in care organisation, we retrospectively evaluated, in a cohort of 700 consecutive probands, the frequency and burden of introducing the search for variants in a maximum list of 244 medically actionable genes (genes that predispose carriers to a preventable or treatable disease in childhood/adulthood and genes for genetic counselling issues). We also focused on the 59 PharmGKB class IA/IB pharmacogenetic variants. We also compared the results in different gene lists. We identified variants (likely) affecting protein function in genes for care in 26 cases (3.7%) and heterozygous variants in genes for genetic counselling in 29 cases (3.8%). Mean time for the 700 patients was about 6.3 min/patient for medically actionable genes and 1.3 min/patient for genes for genetic counselling, and a mean time of 37 min/patients for the reinterpreted variants. These results would lead to all 700 pre-test counselling sessions being longer, to 55 post-test genetic consultations and to 27 secondary specialised medical evaluations. ES also detected 42/59 pharmacogenetic variants or combinations of variants in the majority of cases. An extremely low metabolizer status in genes relevant for neurodevelopmental disorders (CYP2C9 and CYP2C19) was found in 57/700 cases. This study provides information regarding the need to anticipate the implementation of genomic medicine, notably the work overload at various steps of the process.
随着外显子/基因组测序(ES/GS)融入医学实践,报告偶然/次要发现(IFs/SFs)的可能性有所增加。在过去的 4 年中,人们对 IFs/SFs 进行了广泛的研究。为了评估它们对医疗组织的影响,我们回顾性地评估了 700 名连续先证者的队列,这些先证者的频率和负担是在最大的 244 个医学可操作基因(导致携带者在儿童/成年期易患可预防或可治疗疾病的基因和用于遗传咨询问题的基因)中搜索变体。我们还重点研究了 59 个 PharmGKB 类 IA/IB 药物遗传学变体。我们还比较了不同基因列表的结果。我们在 26 例(3.7%)有影响护理相关基因蛋白功能的变异体(可能)和 29 例(3.8%)有遗传咨询相关基因杂合变异体的情况下发现了变异体。700 例患者的平均时间约为 6.3 分钟/例用于医学可操作基因,1.3 分钟/例用于遗传咨询基因,37 分钟/例用于重新解释变异体。这些结果将导致所有 700 例预测试咨询会议时间延长,55 例遗传咨询后咨询,27 例二级专门医疗评估。ES 还在大多数情况下检测到 42/59 个药物遗传学变体或变体组合。在 700 例患者中发现 57 例与神经发育障碍相关的基因(CYP2C9 和 CYP2C19)的极低代谢状态。本研究提供了有关需要预测基因组医学实施的信息,特别是在该过程的各个步骤中工作负载增加的信息。