Singh Aminder A, Shetty Deeti K, Jacob Aishwarya G, Bayraktar Semih, Sinha Sanjay
Wellcome-MRC Cambridge Stem Cell Institute, Jeffrey Cheah Biomedical Centre, Cambridge, United Kingdom.
Front Cardiovasc Med. 2024 Feb 19;11:1349548. doi: 10.3389/fcvm.2024.1349548. eCollection 2024.
Thoracic aortic disease (TAD) is often silent until a life-threatening complication occurs. However, genetic information can inform both identification and treatment at an early stage. Indeed, a diagnosis is important for personalised surveillance and intervention plans, as well as cascade screening of family members. Currently, only 20% of heritable TAD patients have a causative mutation identified and, consequently, further advances in genetic coverage are required to define the remaining molecular landscape. The rapid expansion of next generation sequencing technologies is providing a huge resource of genetic data, but a critical issue remains in functionally validating these findings. Induced pluripotent stem cells (iPSCs) are patient-derived, reprogrammed cell lines which allow mechanistic insights, complex modelling of genetic disease and a platform to study aortic genetic variants. This review will address the need for iPSCs as a frontline diagnostic tool to evaluate variants identified by genomic discovery studies and explore their evolving role in biological insight through to drug discovery.
胸主动脉疾病(TAD)通常在危及生命的并发症出现之前没有症状。然而,基因信息可以在早期阶段为疾病的识别和治疗提供依据。事实上,诊断对于个性化监测和干预计划以及对家庭成员的级联筛查都很重要。目前,只有20%的遗传性TAD患者被确定有致病突变,因此,需要在基因覆盖方面取得进一步进展,以明确其余的分子情况。下一代测序技术的迅速发展提供了大量的基因数据资源,但在功能上验证这些发现仍然是一个关键问题。诱导多能干细胞(iPSC)是源自患者的重编程细胞系,它可以提供机制方面的见解、对遗传疾病进行复杂建模,并为研究主动脉基因变异提供一个平台。本综述将阐述将iPSC作为一线诊断工具的必要性,以评估基因组发现研究中识别出的变异,并探讨其在从生物学洞察到药物发现过程中不断演变的作用。