Santer R, Schmidt-Sommerfeld E, Leung Y K, Fischer J E, Lebenthal E
Department of Paediatrics, Children's Hospital of Buffalo, SUNY.
Eur J Pediatr. 1990 Dec;150(2):111-4. doi: 10.1007/BF02072051.
Inborn errors involving the oxidative metabolism of fatty acids may present clinically with a Reye syndrome-like picture. This case report of a patient with medium-chain acyl CoA dehydrogenase (MCAD) deficiency illustrates that electron microscopy may help to differentiate this disorder from Reye syndrome even if a liver biopsy is performed in a patient who recovered from an acute metabolic decompensation. Together with this case, a review of the few reports in the literature of pathological findings in MCAD deficiency is given. Changes uncharacteristic for Reye syndrome are a large-droplet steatosis and the presence of distinctive mitochondrial abnormalities on electron microscopy. The detection of an electron dense mitochondrial matrix and a widened space of inner mitochondrial membranes rules out Reye syndrome and is suggestive of a disorder of mitochondrial fatty acid oxidation.
涉及脂肪酸氧化代谢的先天性代谢缺陷在临床上可能表现出类似瑞氏综合征的症状。这例中链酰基辅酶A脱氢酶(MCAD)缺乏症患者的病例报告表明,即使在急性代谢失代偿后康复的患者中进行肝活检,电子显微镜检查也可能有助于将这种疾病与瑞氏综合征区分开来。结合该病例,对文献中关于MCAD缺乏症病理结果的少数报告进行了综述。瑞氏综合征不典型的变化是大滴性脂肪变性以及电子显微镜下存在独特的线粒体异常。检测到电子致密的线粒体基质和线粒体内膜间隙增宽可排除瑞氏综合征,并提示线粒体脂肪酸氧化障碍。