Institut de Génomique Fonctionnelle, IGF, CNRS, INSERM, Univ. Montpellier, Montpellier, France.
Research Institute at Nationwide Children's Hospital, Center for Molecular and Human Genetics, Columbus, Ohio, USA.
Stem Cells. 2018 Feb;36(2):192-205. doi: 10.1002/stem.2721. Epub 2017 Nov 10.
One strategy for stem cell-based therapy of the cerebral cortex involves the generation and transplantation of functional, histocompatible cortical-like neurons from embryonic stem cells (ESCs). Diploid parthenogenetic Pg-ESCs have recently emerged as a promising source of histocompatible ESC derivatives for organ regeneration but their utility for cerebral cortex therapy is unknown. A major concern with Pg-ESCs is genomic imprinting. In contrast with biparental Bp-ESCs derived from fertilized oocytes, Pg-ESCs harbor two maternal genomes but no sperm-derived genome. Pg-ESCs are therefore expected to have aberrant expression levels of maternally expressed (MEGs) and paternally expressed (PEGs) imprinted genes. Given the roles of imprinted genes in brain development, tissue homeostasis and cancer, their deregulation in Pg-ESCs might be incompatible with therapy. Here, we report that, unexpectedly, only one gene out of 7 MEGs and 12 PEGs was differentially expressed between Pg-ESCs and Bp-ESCs while 13 were differentially expressed between androgenetic Ag-ESCs and Bp-ESCs, indicating that Pg-ESCs but not Ag-ESCs, have a Bp-like imprinting compatible with therapy. In vitro, Pg-ESCs generated cortical-like progenitors and electrophysiologically active glutamatergic neurons that maintained the Bp-like expression levels for most imprinted genes. In vivo, Pg-ESCs participated to the cortical lineage in fetal chimeras. Finally, transplanted Pg-ESC derivatives integrated into the injured adult cortex and sent axonal projections in the host brain. In conclusion, mouse Pg-ESCs generate functional cortical-like neurons with Bp-like imprinting and their derivatives properly integrate into both the embryonic cortex and the injured adult cortex. Collectively, our data support the utility of Pg-ESCs for cortical therapy. Stem Cells 2018;36:192-205.
一种基于干细胞的大脑皮层治疗策略涉及从胚胎干细胞(ESCs)生成和移植功能性、组织相容性的皮质样神经元。二倍体孤雌生殖 Pg-ESCs 最近成为组织再生的组织相容性 ESC 衍生物的有前途的来源,但它们在大脑皮层治疗中的应用尚不清楚。Pg-ESCs 的一个主要问题是基因组印记。与来自受精卵的双亲 Bp-ESCs 相反,Pg-ESCs 含有两个母体基因组,但没有精子衍生的基因组。因此,预计 Pg-ESCs 会表现出母体表达(MEGs)和父体表达(PEGs)印记基因的异常表达水平。鉴于印记基因在大脑发育、组织稳态和癌症中的作用,它们在 Pg-ESCs 中的失调可能与治疗不兼容。在这里,我们报告出乎意料的是,在 Pg-ESCs 和 Bp-ESCs 之间只有一个基因(7 个 MEGs 和 12 个 PEGs 中的一个)的表达存在差异,而在 Pg-ESCs 和 Bp-ESCs 之间有 13 个基因的表达存在差异,这表明 Pg-ESCs 而不是 Ag-ESCs 具有与治疗相容的 Bp 样印记。在体外,Pg-ESCs 生成皮质样祖细胞和电生理活性谷氨酸能神经元,并保持大多数印记基因的 Bp 样表达水平。在体内,Pg-ESCs 参与胎儿嵌合体的皮质谱系。最后,移植的 Pg-ESC 衍生物整合到损伤的成年皮质中,并在宿主大脑中发出轴突投射。总之,小鼠 Pg-ESCs 生成具有 Bp 样印记的功能性皮质样神经元,其衍生物能够正确整合到胚胎皮质和损伤的成年皮质中。总的来说,我们的数据支持 Pg-ESCs 用于皮质治疗的实用性。干细胞 2018;36:192-205.