Leigh syndrome: Resolving the clinical and genetic heterogeneity paves the way for treatment options

利氏病 遗传异质性 粒线体疾病 呼吸链 线粒体呼吸链 生物信息学 医学 线粒体DNA 基因 神经科学 遗传学 生物 疾病 病理 线粒体 表型
作者
Mike Gerards,Suzanne C.E.H. Sallevelt,Hubert J.M. Smeets
出处
期刊:Molecular Genetics and Metabolism [Elsevier BV]
卷期号:117 (3): 300-312 被引量:131
标识
DOI:10.1016/j.ymgme.2015.12.004
摘要

Leigh syndrome is a progressive neurodegenerative disorder, affecting 1 in 40,000 live births. Most patients present with symptoms between the ages of three and twelve months, but adult onset Leigh syndrome has also been described. The disease course is characterized by a rapid deterioration of cognitive and motor functions, in most cases resulting in death due to respiratory failure. Despite the high genetic heterogeneity of Leigh syndrome, patients present with identical, symmetrical lesions in the basal ganglia or brainstem on MRI, while additional clinical manifestations and age of onset varies from case to case. To date, mutations in over 60 genes, both nuclear and mitochondrial DNA encoded, have been shown to cause Leigh syndrome, still explaining only half of all cases. In most patients, these mutations directly or indirectly affect the activity of the mitochondrial respiratory chain or pyruvate dehydrogenase complex. Exome sequencing has accelerated the discovery of new genes and pathways involved in Leigh syndrome, providing novel insights into the pathophysiological mechanisms. This is particularly important as no general curative treatment is available for this devastating disorder, although several recent studies imply that early treatment might be beneficial for some patients depending on the gene or process affected. Timely, gene-based personalized treatment may become an important strategy in rare, genetically heterogeneous disorders like Leigh syndrome, stressing the importance of early genetic diagnosis and identification of new genes/pathways. In this review, we provide a comprehensive overview of the most important clinical manifestations and genes/pathways involved in Leigh syndrome, and discuss the current state of therapeutic interventions in patients.
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