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对疑似线粒体疾病的深入评估。

The in-depth evaluation of suspected mitochondrial disease.

作者信息

Haas Richard H, Parikh Sumit, Falk Marni J, Saneto Russell P, Wolf Nicole I, Darin Niklas, Wong Lee-Jun, Cohen Bruce H, Naviaux Robert K

机构信息

Department of Neurosciences, University of California San Diego, La Jolla, CA and Rady Children's Hospital San Diego, San Diego, CA, United States.

出版信息

Mol Genet Metab. 2008 May;94(1):16-37. doi: 10.1016/j.ymgme.2007.11.018. Epub 2008 Feb 1.

Abstract

Mitochondrial disease confirmation and establishment of a specific molecular diagnosis requires extensive clinical and laboratory evaluation. Dual genome origins of mitochondrial disease, multi-organ system manifestations, and an ever increasing spectrum of recognized phenotypes represent the main diagnostic challenges. To overcome these obstacles, compiling information from a variety of diagnostic laboratory modalities can often provide sufficient evidence to establish an etiology. These include blood and tissue histochemical and analyte measurements, neuroimaging, provocative testing, enzymatic assays of tissue samples and cultured cells, as well as DNA analysis. As interpretation of results from these multifaceted investigations can become quite complex, the Diagnostic Committee of the Mitochondrial Medicine Society developed this review to provide an overview of currently available and emerging methodologies for the diagnosis of primary mitochondrial disease, with a focus on disorders characterized by impairment of oxidative phosphorylation. The aim of this work is to facilitate the diagnosis of mitochondrial disease by geneticists, neurologists, and other metabolic specialists who face the challenge of evaluating patients of all ages with suspected mitochondrial disease.

摘要

线粒体疾病的确证及特定分子诊断的建立需要广泛的临床和实验室评估。线粒体疾病的双重基因组起源、多器官系统表现以及不断增加的已识别表型谱构成了主要的诊断挑战。为克服这些障碍,整合来自各种诊断实验室检测方法的信息通常能够提供足够的证据来确定病因。这些方法包括血液和组织的组织化学及分析物测量、神经影像学检查、激发试验、组织样本和培养细胞的酶活性测定以及DNA分析。由于对这些多方面调查结果的解读可能会变得相当复杂,线粒体医学协会诊断委员会编写了本综述,以概述目前用于诊断原发性线粒体疾病的现有方法和新兴方法,重点关注以氧化磷酸化受损为特征的疾病。这项工作的目的是帮助遗传学家、神经学家和其他代谢专家诊断线粒体疾病,他们在评估各年龄段疑似线粒体疾病患者时面临挑战。

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