Yu Ruofan, Roseman Shelby, Siegenfeld Allison P, Nguyen Son C, Joyce Eric F, Liau Brian B, Krantz Ian D, Alexander Katherine A, Berger Shelley L
bioRxiv. 2023 Oct 24:2023.07.22.550178. doi: 10.1101/2023.07.22.550178.
The interchromatin space in the cell nucleus contains various membrane-less nuclear bodies. Recent findings indicate that nuclear speckles, comprising a distinct nuclear body, exhibit interactions with certain chromatin regions in a ground state. Key questions are how this ground state of chromatin-nuclear speckle association is established and what are the gene regulatory roles of this layer of nuclear organization. We report here that chromatin structural factors CTCF and cohesin are required for full ground state association between DNA and nuclear speckles. Disruption of ground state DNA-speckle contacts via either CTCF depletion or cohesin depletion had minor effects on basal level expression of speckle-associated genes, however we show strong negative effects on stimulus-dependent induction of speckle-associated genes. We identified a putative speckle targeting motif (STM) within cohesin subunit RAD21 and demonstrated that the STM is required for chromatin-nuclear speckle association. In contrast to reduction of CTCF or RAD21, depletion of the cohesin releasing factor WAPL stabilized cohesin on chromatin and DNA-speckle contacts, resulting in enhanced inducibility of speckle-associated genes. In addition, we observed disruption of chromatin-nuclear speckle association in patient derived cells with Cornelia de Lange syndrome (CdLS), a congenital neurodevelopmental diagnosis involving defective cohesin pathways, thus revealing nuclear speckles as an avenue for therapeutic inquiry. In summary, our findings reveal a mechanism to establish the ground organizational state of chromatin-speckle association, to promote gene inducibility, and with relevance to human disease.
细胞核中的染色质间空间包含各种无膜核体。最近的研究结果表明,作为一种独特核体的核斑点在基态下与某些染色质区域存在相互作用。关键问题在于这种染色质 - 核斑点关联的基态是如何建立的,以及这一层核组织的基因调控作用是什么。我们在此报告,染色质结构因子CTCF和黏连蛋白是DNA与核斑点之间完全基态关联所必需的。通过CTCF缺失或黏连蛋白缺失破坏基态DNA - 斑点接触,对斑点相关基因的基础水平表达影响较小,但我们发现对斑点相关基因的刺激依赖性诱导有强烈的负面影响。我们在黏连蛋白亚基RAD21中鉴定出一个假定的斑点靶向基序(STM),并证明STM是染色质 - 核斑点关联所必需 的。与CTCF或RAD21减少相反,黏连蛋白释放因子WAPL的缺失使黏连蛋白稳定在染色质和DNA - 斑点接触上,导致斑点相关基因的诱导性增强。此外,我们在患有科妮莉亚·德朗格综合征(CdLS)的患者来源细胞中观察到染色质 - 核斑点关联的破坏,CdLS是一种涉及黏连蛋白途径缺陷的先天性神经发育疾病,从而揭示核斑点是一个治疗研究的途径。总之,我们的研究结果揭示了一种建立染色质 - 斑点关联的基础组织状态、促进基因诱导性并与人类疾病相关的机制。