The PXE International Center of Excellence in Research and Clinical Care, Department of Dermatology and Cutaneous Biology, The Sidney Kimmel Medical College, Thomas Jefferson University, Philadelphia, PA, USA; Department of Dermatology, The First Affiliated Hospital of Zhejiang Chinese Medical University, Hangzhou, Zhejiang, China.
The PXE International Center of Excellence in Research and Clinical Care, Department of Dermatology and Cutaneous Biology, The Sidney Kimmel Medical College, Thomas Jefferson University, Philadelphia, PA, USA.
J Invest Dermatol. 2021 May;141(5):1148-1156. doi: 10.1016/j.jid.2020.10.013. Epub 2020 Dec 17.
In the past two decades, there has been great progress in identifying the molecular basis and pathomechanistic details in pseudoxanthoma elasticum (PXE), a heritable multisystem ectopic mineralization disorder. Although the identification of pathogenic variants in ABCC6 has been critical for understanding the disease process, genetic modifiers have been disclosed that explain the phenotypic heterogeneity of PXE. Adding to the genetic complexity of PXE are PXE-like phenotypes caused by pathogenic variants in other ectopic mineralization-associated genes. This review summarizes the current knowledge of the genetics and candidate modifier genes in PXE, a multifactorial disease at the genome-environment interface.
在过去的二十年中,人们在识别弹性假黄瘤(PXE)的分子基础和发病机制细节方面取得了重大进展,PXE 是一种遗传性多系统异位矿化紊乱。尽管 ABCC6 中的致病变异的鉴定对于了解疾病过程至关重要,但已经揭示了遗传修饰因子,这些因子解释了 PXE 的表型异质性。使 PXE 的遗传复杂性增加的是由其他异位矿化相关基因中的致病变异引起的 PXE 样表型。这篇综述总结了 PXE 遗传和候选修饰基因的最新知识,PXE 是基因组-环境界面的一种多因素疾病。