Michigan Neuroscience Institute (MNI), University of Michigan, Ann Arbor, MI, 48109, USA.
Department of Human Genetics, University of Michigan, Ann Arbor, MI, 48109, USA.
Nat Commun. 2020 Jul 31;11(1):3839. doi: 10.1038/s41467-020-17551-4.
Chromatin regulates spatiotemporal gene expression during neurodevelopment, but it also mediates DNA damage repair essential to proliferating neural progenitor cells (NPCs). Here, we uncover molecularly dissociable roles for nucleosome remodeler Ino80 in chromatin-mediated transcriptional regulation and genome maintenance in corticogenesis. We find that conditional Ino80 deletion from cortical NPCs impairs DNA double-strand break (DSB) repair, triggering p53-dependent apoptosis and microcephaly. Using an in vivo DSB repair pathway assay, we find that Ino80 is selectively required for homologous recombination (HR) DNA repair, which is mechanistically distinct from Ino80 function in YY1-associated transcription. Unexpectedly, sensitivity to loss of Ino80-mediated HR is dependent on NPC division mode: Ino80 deletion leads to unrepaired DNA breaks and apoptosis in symmetric NPC-NPC divisions, but not in asymmetric neurogenic divisions. This division mode dependence is phenocopied following conditional deletion of HR gene Brca2. Thus, distinct modes of NPC division have divergent requirements for Ino80-dependent HR DNA repair.
染色质在神经发育过程中调节时空基因表达,但它也介导增殖性神经祖细胞(NPC)中必需的 DNA 损伤修复。在这里,我们揭示了核小体重塑酶 Ino80 在皮质发生中染色质介导的转录调控和基因组维持方面具有分子可分离的作用。我们发现,条件性 Ino80 缺失皮质 NPC 会损害 DNA 双链断裂(DSB)修复,触发 p53 依赖性细胞凋亡和小头畸形。使用体内 DSB 修复途径测定法,我们发现 Ino80 选择性地需要同源重组(HR)DNA 修复,其机制与 Ino80 在 YY1 相关转录中的功能不同。出乎意料的是,对 Ino80 介导的 HR 缺失的敏感性取决于 NPC 分裂模式:Ino80 缺失导致对称 NPC-NPC 分裂中未修复的 DNA 断裂和细胞凋亡,但在不对称神经发生分裂中则不会。在条件性 HR 基因 Brca2 缺失后,出现了这种分裂模式依赖性。因此,NPC 分裂的不同模式对依赖 Ino80 的 HR DNA 修复具有不同的要求。