Institute of Biomembranes and Bioenergetics, National Council of Research, Bari, Italy.
Section of Behavioural Neuroscience, Department of Cell Biology and Neurosciences, Istituto Superiore di Sanità Roma, Italy.
Neurosci Biobehav Rev. 2014 Oct;46 Pt 2:202-17. doi: 10.1016/j.neubiorev.2014.01.012. Epub 2014 Feb 15.
Clinical manifestations typical of mitochondrial diseases are often present in various genetic syndromes associated with intellectual disability, a condition leading to deficit in cognitive functions and adaptive behaviors. Until now, the causative mechanism leading to intellectual disability is unknown and the progression of the condition is poorly understood. We first report latest advances on genetic and environmental regulation of mitochondrial function and its role in brain development. Starting from the structure, function and regulation of the oxidative phosphorylation apparatus, we review how mitochondrial biogenesis and dynamics play a central role in neurogenesis and neuroplasticity. We then discuss how dysfunctional mitochondria and alterations in reactive oxygen species homeostasis are potentially involved in the pathogenesis of various neurodevelopmental syndromes with a special focus on Down, Rett, Fragile X syndromes and autism spectrum disorders. Finally, we review and suggest novel therapeutic approaches aimed at improving intellectual disability by activating mitochondrial function and reducing oxidative stress to amiliorate the quality of life in the subjects affected.
线粒体疾病的临床表现通常存在于各种与智力障碍相关的遗传综合征中,这种疾病导致认知功能和适应行为的缺陷。到目前为止,导致智力障碍的发病机制尚不清楚,病情的进展也知之甚少。我们首先报告了线粒体功能的遗传和环境调控及其在大脑发育中的作用的最新进展。从氧化磷酸化装置的结构、功能和调节开始,我们回顾了线粒体生物发生和动力学如何在神经发生和神经可塑性中发挥核心作用。然后,我们讨论了功能失调的线粒体和活性氧物质稳态的改变如何可能参与各种神经发育综合征的发病机制,特别关注唐氏综合征、雷特综合征、脆性 X 综合征和自闭症谱系障碍。最后,我们回顾并提出了新的治疗方法,旨在通过激活线粒体功能和减少氧化应激来改善智力障碍,从而改善受影响个体的生活质量。