Enkai Shigehiro, Koinuma Sachi, Ito Reiko, Igaki Junko, Hasegawa Yukihiro, Murayama Kei, Ohtake Akira
Department of Pediatrics, Fussa Hospital, Tokyo, Japan.
Pediatr Int. 2013 Aug;55(4):e103-6. doi: 10.1111/ped.12098.
The patient had hepatomegaly with liver dysfunction at the age of 1 month. Magnetic resonance imaging performed at the age of 1 year showed multiple nodules of varying size in his liver. We were able to examine the mitochondrial respiratory chain function in the liver biopsy samples because all other differential diagnoses for hepatic cirrhosis had been ruled out. Complex I and IV activities were below the normal level (<30%) of the citrate synthase (CS) ratio. Liver blue native polyacrylamide gel electrophoresis showed an extremely weak complex I and IV band. Liver respiratory chain complexes I and IV were found to be deficient in this patient. The histologic findings were highly suggestive of mitochondrial respiratory chain disorder. Findings of progressive liver cirrhosis changes were observed in magnetic resonance imaging at the age of 5 years. An examination of the mitochondrial respiratory chain function should be performed along with a liver biopsy if mitochondrial respiratory chain disorder is suspected as a possible differential diagnosis of idiopathic hepatitis.
该患者1个月大时出现肝肿大并伴有肝功能障碍。1岁时进行的磁共振成像显示其肝脏中有多个大小不一的结节。由于已排除肝硬化的所有其他鉴别诊断,我们能够对肝活检样本中的线粒体呼吸链功能进行检测。复合体I和IV的活性低于柠檬酸合酶(CS)比率的正常水平(<30%)。肝脏蓝色天然聚丙烯酰胺凝胶电泳显示复合体I和IV条带极其微弱。该患者肝脏呼吸链复合体I和IV存在缺陷。组织学检查结果高度提示线粒体呼吸链疾病。5岁时的磁共振成像观察到进行性肝硬化改变。如果怀疑线粒体呼吸链疾病可能是特发性肝炎的鉴别诊断之一,则应在进行肝活检的同时检查线粒体呼吸链功能。