INSERM, U968, Paris, 75012, France.
J Inherit Metab Dis. 2011 Apr;34(2):327-44. doi: 10.1007/s10545-010-9131-5. Epub 2010 Jun 23.
Mitochondrial disorders can no longer be ignored in most medical disciplines. Such disorders include specific and widespread organ involvement, with tissue degeneration or tumor formation. Primary or secondary actors, mitochondrial dysfunctions also play a role in the aging process. Despite progresses made in identification of their molecular bases, nearly everything remains to be done as regards therapy. Research dealing with mitochondrial physiology and pathology has >20 years of history around the world. We are involved, as are many other laboratories, in the challenge of finding ways to fight these diseases. However, our main limitation is the scarcety of animal models required for both understanding the molecular mechanisms underlying the diseases and evaluating therapeutic strategies. This is especially true for diseases due to mutations in mitochondrial DNA (mtDNA), since an authentic genetic model of mtDNA mutations is technically a very difficult task due to both the inability of manipulating the mitochondrial genome of living mammalian cells and to its multicopy nature. This has led researchers in the field to consider the prospect of gene therapy approaches that can roughly be divided into three groups: (1) import of wild-type copies or relevant sections of DNA or RNA into mitochondria, (2) manipulation of mitochondrial genetic content, and (3) rescue of a defect by expression of an engineered gene product from the nucleus (allotopic or xenotropic expression). We briefly introduce these concepts and indicate where promising progress has been made in the last decade.
线粒体疾病在大多数医学领域已不容忽视。这些疾病包括特定和广泛的器官受累,伴有组织退化或肿瘤形成。原发性或继发性因素,线粒体功能障碍也在衰老过程中起作用。尽管在鉴定其分子基础方面取得了进展,但几乎在治疗方面仍有许多工作要做。涉及线粒体生理学和病理学的研究在全球已有 20 多年的历史。我们与许多其他实验室一起,参与了寻找对抗这些疾病的方法的挑战。然而,我们的主要限制是需要大量的动物模型,既要了解疾病相关的分子机制,又要评估治疗策略。对于由线粒体 DNA (mtDNA) 突变引起的疾病来说尤其如此,因为由于无法操纵活哺乳动物细胞的线粒体基因组及其多拷贝性质,因此,真正的 mtDNA 突变的遗传模型在技术上是一项非常困难的任务。这导致该领域的研究人员考虑采用基因治疗方法,这些方法大致可以分为三组:(1)将野生型拷贝或 DNA 或 RNA 的相关部分导入线粒体,(2)操纵线粒体遗传物质,以及(3)通过从细胞核表达工程基因产物(异位或异源表达)来挽救缺陷。我们简要介绍了这些概念,并指出了在过去十年中取得了有希望进展的地方。