Bloor Stuart, Wit Niek, Lehner Paul J
Cambridge Institute of Therapeutic Immunology & Infectious Disease (CITIID), Jeffrey Cheah Biomedical Centre, Department of Medicine, University of Cambridge, Cambridge, CB2 0AW, UK.
Nucleic Acids Res. 2025 Jan 11;53(2). doi: 10.1093/nar/gkae1165.
The human silencing hub (HUSH) complex is a transcription-dependent, epigenetic repressor complex that provides a genome-wide immunosurveillance system for the recognition and silencing of newly-integrated retroelements. The core HUSH complex of TASOR, MPP8 and Periphilin, represses these retroelements through SETDB1-mediated H3K9me3 deposition and MORC2-dependent chromatin compaction. HUSH-dependent silencing is RNA-mediated, yet no HUSH component contains a recognised RNA-binding domain. Here we used an unbiased approach to identify which HUSH component was able to bind RNA and determine whether RNA-binding was essential for HUSH function. We identify Periphilin as the major RNA-binding component of the HUSH complex and show that Periphilin's N-terminal domain is essential for both RNA binding and HUSH function. Periphilin binding to RNA was independent of its interaction with TASOR or MPP8, as its N-terminal domain was sufficient for RNA targeting. The artificial tethering of Periphilin to a HUSH-insensitive, nascent transcript, enabled the HUSH-dependent silencing of the transcript. This tethering of Periphilin allowed the RNA-binding region of Periphilin to be removed such that only its C-terminal domain was required for oligomerisation and interaction with TASOR. We therefore show that Periphilin is the predominant RNA-binding protein of the HUSH complex and this RNA-binding is essential for HUSH activity.
人类沉默枢纽(HUSH)复合体是一种转录依赖性的表观遗传抑制复合体,它提供了一种全基因组免疫监视系统,用于识别和沉默新整合的逆转录元件。由TASOR、MPP8和外周素组成的核心HUSH复合体,通过SETDB1介导的H3K9me3沉积和MORC2依赖性染色质压缩来抑制这些逆转录元件。HUSH依赖性沉默是由RNA介导的,但HUSH复合体的任何组分都不包含公认的RNA结合结构域。在这里,我们采用了一种无偏向性的方法来确定HUSH复合体的哪个组分能够结合RNA,并确定RNA结合对于HUSH功能是否至关重要。我们确定外周素是HUSH复合体的主要RNA结合组分,并表明外周素的N端结构域对于RNA结合和HUSH功能均至关重要。外周素与RNA的结合独立于其与TASOR或MPP8的相互作用,因为其N端结构域足以实现RNA靶向。将外周素人工拴系到对HUSH不敏感的新生转录本上,能够使该转录本发生HUSH依赖性沉默。外周素的这种拴系使得外周素的RNA结合区域可以被去除,这样寡聚化以及与TASOR相互作用仅需要其C端结构域。因此,我们表明外周素是HUSH复合体的主要RNA结合蛋白,并且这种RNA结合对于HUSH活性至关重要。