Verdile Veronica, De Paola Elisa, Paronetto Maria Paola
University of Rome "Foro Italico", Rome, Italy.
Laboratory of Cellular and Molecular Neurobiology, Fondazione Santa Lucia, Rome, Italy.
Front Genet. 2019 Mar 22;10:173. doi: 10.3389/fgene.2019.00173. eCollection 2019.
Phase separation is a physiological process occurring spontaneously when single-phase molecular complexes separate in two phases, a concentrated phase and a more diluted one. Eukaryotic cells employ phase transition strategies to promote the formation of intracellular territories not delimited by membranes with increased local RNA concentration, such as nucleolus, paraspeckles, P granules, Cajal bodies, P-bodies, and stress granules. These organelles contain both proteins and coding and non-coding RNAs and play important roles in different steps of the regulation of gene expression and in cellular signaling. Recently, it has been shown that most human RNA-binding proteins (RBPs) contain at least one low-complexity domain, called prion-like domain (PrLD), because proteins harboring them display aggregation properties like prion proteins. PrLDs support RBP function and contribute to liquid-liquid phase transitions that drive ribonucleoprotein granule assembly, but also render RBPs prone to misfolding by promoting the formation of pathological aggregates that lead to toxicity in specific cell types. Protein-protein and protein-RNA interactions within the separated phase can enhance the transition of RBPs into solid aberrant aggregates, thus causing diseases. In this review, we highlight the role of phase transition in human disease such as amyotrophic lateral sclerosis (ALS), frontotemporal dementia (FTD), and in cancer. Moreover, we discuss novel therapeutic strategies focused to control phase transitions by preventing the conversion into aberrant aggregates. In this regard, the stimulation of chaperone machinery to disassemble membrane-less organelles, the induction of pathways that could inhibit aberrant phase separation, and the development of antisense oligonucleotides (ASOs) to knockdown RNAs could be evaluated as novel therapeutic strategies for the treatment of those human diseases characterized by aberrant phase transition aggregates.
相分离是一种生理过程,当单相分子复合物分离为两个相,即浓缩相和更稀释的相时自发发生。真核细胞采用相变策略来促进形成细胞内区域,这些区域不由膜界定,但局部RNA浓度增加,如核仁、副斑点、P小体、卡哈尔体、P体和应激颗粒。这些细胞器包含蛋白质以及编码和非编码RNA,并在基因表达调控的不同步骤和细胞信号传导中发挥重要作用。最近的研究表明,大多数人类RNA结合蛋白(RBP)至少包含一个低复杂性结构域,称为朊病毒样结构域(PrLD),因为含有这些结构域的蛋白质表现出类似朊病毒蛋白的聚集特性。PrLD支持RBP功能,并有助于驱动核糖核蛋白颗粒组装的液-液相转变,但也会通过促进病理性聚集体的形成使RBP易于错误折叠,从而在特定细胞类型中导致毒性。分离相中蛋白质-蛋白质和蛋白质-RNA相互作用可增强RBP向固体异常聚集体的转变,从而引发疾病。在这篇综述中,我们强调了相变在人类疾病如肌萎缩侧索硬化症(ALS)、额颞叶痴呆(FTD)和癌症中的作用。此外,我们讨论了旨在通过防止转化为异常聚集体来控制相变的新型治疗策略。在这方面,刺激伴侣机制以拆解无膜细胞器、诱导可抑制异常相分离的途径以及开发反义寡核苷酸(ASO)以敲低RNA可作为治疗那些以异常相变聚集体为特征的人类疾病的新型治疗策略进行评估。