Sampson Matthew G
Division of Pediatric Nephrology, Department of Pediatrics and Communicable Diseases, University of Michigan School of Medicine, Ann Arbor, Michigan, United States.
J Pediatr Genet. 2016 Mar;5(1):69-75. doi: 10.1055/s-0035-1557113. Epub 2015 Aug 13.
The discovery of genetic variation associated with pediatric kidney disease has shed light on the biology underlying these conditions and, in some cases, has improved our clinical management of patients. We are challenged to continue the momentum of the genomic era in pediatric nephrology by identifying novel disease-associated genetic variation and translating these discoveries into clinical applications. This article reviews the diverse forms of genetic architecture that have been found to be associated with kidney diseases and traits. These include rare, fully penetrant variants responsible for Mendelian forms of disease, copy number variants, and more common variants associated with increased risk of disease. These discoveries have provided us with a greater understanding of the molecular mechanisms underlying these conditions and highlighted key pathways for potential intervention. In a number of areas, the identification of rare, fully penetrant variants is immediately clinically relevant, whether in regard to diagnostic testing, prediction of outcomes, or choice of therapies and interventions. This article discusses limitations in the deterministic view of rare, putatively causal mutations, a challenge increasing in importance as sequencing expands to many more genes and patients. This article also focusses on common genetic variants, using those found to be associated with focal segmental glomerulosclerosis in African-Americans, IgA nephropathy, chronic kidney disease (CKD), and estimated glomerular filtration rate (eGFR) as examples. Identifying common genetic variants associated with disease will complement other areas of genomic inquiry, lead to a greater biological understanding of disease, and will benefit pediatric nephrology patients.
与小儿肾脏疾病相关的基因变异的发现,揭示了这些疾病背后的生物学机制,并且在某些情况下,改善了我们对患者的临床管理。我们面临的挑战是,通过识别新的疾病相关基因变异并将这些发现转化为临床应用,来延续小儿肾脏病学基因组时代的发展势头。本文综述了已发现的与肾脏疾病和特征相关的多种基因结构形式。这些包括导致孟德尔式疾病的罕见、完全显性的变异、拷贝数变异,以及与疾病风险增加相关的更常见变异。这些发现使我们对这些疾病背后的分子机制有了更深入的了解,并突出了潜在干预的关键途径。在许多领域,罕见、完全显性变异的识别在临床上具有直接相关性,无论是在诊断测试、预后预测还是治疗和干预的选择方面。本文讨论了对罕见的、假定为因果关系的突变的确定性观点的局限性,随着测序扩展到更多基因和患者,这一挑战的重要性日益增加。本文还聚焦于常见基因变异,以在非裔美国人中发现的与局灶节段性肾小球硬化、IgA肾病、慢性肾脏病(CKD)和估计肾小球滤过率(eGFR)相关的变异为例。识别与疾病相关的常见基因变异将补充基因组研究的其他领域,加深对疾病的生物学理解,并造福小儿肾脏病患者。