Stanley C A, Hale D E
University of Pennsylvania School of Medicine, Philadelphia.
Curr Opin Pediatr. 1994 Aug;6(4):476-81. doi: 10.1097/00008480-199408000-00021.
Genetic diseases of mitochondrial fatty acid oxidation have recently emerged as important disorders to consider in the differential diagnosis of hypoglycemia, cardiomyopathy, or skeletal muscle weakness in infants and children. A total of 16 different defects have been identified over the past decade that involve almost all of the possible enzyme steps in the pathway. One of these disorders, medium-chain acyl-coenzyme A dehydrogenase deficiency has a frequency as high as 1 in 10,000 births and is the single most common genetic defect of intermediary metabolism. The disorders are frequently mistaken for Reye syndrome or sudden infant death syndrome. Improved methods have simplified the diagnosis of some of the fatty acid oxidation defects. However, recognition of these disorders remains challenging. Rapid advances have continued to be made over the past year in defining clinical phenotypes, diagnostic methods, and therapeutic strategies. Familiarity with this new group of disorders is becoming increasingly important for general pediatricians as well as subspecialists in metabolism, endocrinology, gastroenterology, cardiology, neurology, and genetics.
线粒体脂肪酸氧化的遗传疾病最近已成为婴幼儿低血糖、心肌病或骨骼肌无力鉴别诊断中需考虑的重要疾病。在过去十年中,总共发现了16种不同的缺陷,几乎涉及该途径中所有可能的酶促步骤。其中一种疾病,即中链酰基辅酶A脱氢酶缺乏症,在出生婴儿中的发病率高达万分之一,是中间代谢中最常见的单一遗传缺陷。这些疾病常被误诊为瑞氏综合征或婴儿猝死综合征。改进的方法简化了一些脂肪酸氧化缺陷的诊断。然而,识别这些疾病仍然具有挑战性。在过去一年里,在确定临床表型、诊断方法和治疗策略方面继续取得快速进展。对于普通儿科医生以及代谢、内分泌、胃肠病、心脏病、神经学和遗传学领域的专科医生来说,熟悉这一组新疾病变得越来越重要。