Lee H F, Mak S C, Wu F W, Chi C S, Huang S C
Department of Pediatrics, Taichung Veterans General Hospital, 160, Chung Kang Road, Sec. 3, Taichung, Taiwan.
Acta Paediatr Taiwan. 2001 Jan-Feb;42(1):53-6.
Zellweger syndrome is a fatal autosomal-recessive hereditary disease characterized by the absence of peroxisomes in liver and kidneys. The absence of peroxisomes results in impairment of many metabolic pathways, especially beta-oxidation of very long chain fatty acids (VLCFAs). We report a case of a three-month-old male infant with facial dysmorphism, hypotonia, psychomotor retardation, and hepatomegaly. He had an elder brother with the same facial features and hypotonia who died of hepatic failure at four months of age. Biochemical studies revealed elevation of blood pipecolic acid and VLCFAs, compatible with peroxisomal disorder. Electron microscopy of liver biopsy revealed absence of peroxisomes. Zellweger syndrome was diagnosed. Because this syndrome is usually fatal in early life, genetic counseling and prenatal diagnosis are crucial.
脑肝肾综合征是一种致命的常染色体隐性遗传病,其特征是肝脏和肾脏中缺乏过氧化物酶体。过氧化物酶体的缺失导致许多代谢途径受损,尤其是极长链脂肪酸(VLCFAs)的β氧化。我们报告一例三个月大的男婴,有面部畸形、肌张力减退、精神运动发育迟缓及肝肿大。他有一个哥哥,面部特征相同且有肌张力减退,在四个月大时死于肝功能衰竭。生化研究显示血中哌可酸和VLCFAs升高,符合过氧化物酶体疾病。肝活检的电子显微镜检查显示过氧化物酶体缺失。诊断为脑肝肾综合征。由于该综合征在生命早期通常是致命的,遗传咨询和产前诊断至关重要。