Genetics and Genome Biology Program, The Hospital for Sick Children, The Peter Gilgan Centre for Research and Learning, Toronto, ON, M5G 0A4, Canada.
Department of Molecular Genetics, University of Toronto, Toronto, ON, M5S 1A1, Canada.
Mamm Genome. 2019 Jun;30(5-6):90-110. doi: 10.1007/s00335-019-09793-5. Epub 2019 Feb 28.
Rare diseases are very difficult to study mechanistically and to develop therapies for because of the scarcity of patients. Here, the rare neuro-metabolic disorder Rett syndrome (RTT) is discussed as a prototype for precision medicine, demonstrating how mouse models have led to an understanding of the development of symptoms. RTT is caused by mutations in the X-linked gene methyl-CpG-binding protein 2 (MECP2). Mecp2-mutant mice are being used in preclinical studies that target the MECP2 gene directly, or its downstream pathways. Importantly, this work may improve the health of RTT patients. Clinical presentation may vary widely among individuals based on their mutation, but also because of the degree of X chromosome inactivation and the presence of modifier genes. Because it is a complex disorder involving many organ systems, it is likely that recovery of RTT patients will involve a combination of treatments. Precision medicine is warranted to provide the best efficacy to individually treat RTT patients.
罕见病由于患者数量稀少,在机制研究和治疗方法开发方面都极具挑战。本文以神经代谢罕见病雷特综合征(RTT)为例,讨论精准医学的应用,阐明小鼠模型如何帮助我们理解疾病症状的发展。RTT 是由 X 连锁基因甲基化CpG 结合蛋白 2(MECP2)突变引起的。目前,已有研究利用 Mecp2 突变小鼠直接针对 MECP2 基因或其下游通路开展临床前研究。重要的是,这一工作可能会改善 RTT 患者的健康状况。基于其突变情况以及 X 染色体失活程度和修饰基因的存在,个体的临床表现可能存在广泛差异。由于 RTT 是一种涉及多个器官系统的复杂疾病,RTT 患者的康复很可能需要多种治疗方法的结合。因此,有必要采用精准医学方法,为 RTT 患者提供最佳的个体化治疗效果。