Bennett M J, Rinaldo P, Strauss A W
Department of Pathology, University of Texas Southwestern Medical Center, Dallas 75235, USA.
Crit Rev Clin Lab Sci. 2000 Feb;37(1):1-44. doi: 10.1080/10408360091174169.
Inborn errors of the mitochondrial beta-oxidation of long-chain fatty acids represent an evolving field of inherited metabolic disease. Fatty acid oxidation defects demonstrate an abnormal response to the process of fasting adaptation and affect those tissues that utilize fatty acids as an energy source. These tissues include cardiac and skeletal muscle and liver. Muscle directly uses fatty acids as an energy source whilst hepatic metabolism of fatty acids is mostly directed toward the synthesis of ketone bodies for energy utilization by tissues such as brain. The clinical phenotypes of fatty acid oxidation disorders include disease of one or more of these fatty acid-metabolizing tissues. In this review, we provide an overview of the pathway, discuss the disorders that are well established, and describe recent advances in the field. Currently available diagnostic procedures are critically evaluated.
长链脂肪酸线粒体β氧化的先天性缺陷是遗传性代谢疾病中一个不断发展的领域。脂肪酸氧化缺陷表现出对禁食适应过程的异常反应,并影响那些将脂肪酸作为能量来源的组织。这些组织包括心肌、骨骼肌和肝脏。肌肉直接将脂肪酸用作能量来源,而肝脏对脂肪酸的代谢主要是合成酮体,以供大脑等组织用于能量利用。脂肪酸氧化障碍的临床表型包括这些脂肪酸代谢组织中一种或多种的疾病。在本综述中,我们概述了该途径,讨论了已明确的疾病,并描述了该领域的最新进展。对目前可用的诊断程序进行了严格评估。