Joint School of Life Sciences, Guangzhou Institutes of Biomedicine and Health, Chinese Academy of Sciences; Guangzhou Medical University, Guangzhou, 510530, China.
Centre for Regenerative Medicine and Health, Hong Kong Institute of Science & Innovation, Chinese Academy of Sciences, Hong Kong, 99077, China.
Sci China Life Sci. 2024 Nov;67(11):2511-2519. doi: 10.1007/s11427-024-2647-8. Epub 2024 Aug 8.
Leber's hereditary optic neuropathy (LHON) is a debilitating mitochondrial disease associated with mutations in mitochondrial DNA (mtDNA). Unfortunately, the available treatment options for LHON patients are limited due to challenges in mitochondrial replacement. In our study, we reprogramming LHON urine cells into induced pluripotent stem cells (iPSCs) and differentiating them into neural progenitor cells (NPCs) and neurons for disease modeling. Our research revealed that LHON neurons exhibited significantly higher levels of mtDNA mutations and reduced mitochondrial function, confirming the disease phenotype. However, through co-culturing LHON iPSC-derived NPCs with mesenchymal stem cells (MSCs), we observed a remarkable rescue of mutant mtDNA and a significant improvement in mitochondrial metabolic function in LHON neurons. These findings suggest that co-culturing with MSCs can enhance mitochondrial function in LHON NPCs, even after their differentiation into neurons. This discovery holds promise as a potential therapeutic strategy for LHON patients.
Leber 遗传性视神经病变(LHON)是一种与线粒体 DNA(mtDNA)突变相关的使人虚弱的线粒体疾病。不幸的是,由于线粒体替代存在挑战,LHON 患者的可用治疗选择有限。在我们的研究中,我们将 LHON 尿液细胞重编程为诱导多能干细胞(iPSC),并将其分化为神经祖细胞(NPC)和神经元以进行疾病建模。我们的研究表明,LHON 神经元表现出明显更高水平的 mtDNA 突变和线粒体功能降低,证实了疾病表型。然而,通过将 LHON iPSC 衍生的 NPC 与间充质干细胞(MSC)共培养,我们观察到突变型 mtDNA 的显著挽救和 LHON 神经元中线粒体代谢功能的显著改善。这些发现表明,与 MSC 共培养可以增强 LHON NPC 中的线粒体功能,即使在它们分化为神经元之后也是如此。这一发现为 LHON 患者提供了一种有潜力的治疗策略。