瓜氨酸血症
医学
复合杂合度
无义突变
未能茁壮成长
突变
胆汁淤积
高氨血症
血脂异常
内科学
新生儿肝炎
胃肠病学
儿科
错义突变
遗传学
尿素循环
基因
生物
胆道闭锁
肝移植
移植
氨基酸
精氨酸
肥胖
作者
Daniela Avdjieva-Tzavella,M. B. Ivanova,Tihomir Todorov,Albena Todorova,E I Panteleeva,Savina Tincheva,E A Lazarova,Hadil Kathom,P G Yaneva,Radka Tincheva
出处
期刊:Genetic Counseling
日期:2014-01-01
卷期号:25 (3): 271-6
被引量:6
摘要
Citrin deficiency is an autosomal recessive disorder caused by mutations in the SLC25A13 gene and has three phenotypes: neonatal intrahepatic cholestasis caused by citrin deficiency (NICCD) in newborns, failure to thrive and dyslipidemia caused by citrin deficiency (FTTDCD) in older children, and recurrent hyperammonemia with neuropsychiatric symptoms in citrullinemia type II (CTLN2) in adults. NICCD presents in the first few weeks of life with cholestatic hepatitis syndrome, multiple aminoacidemia and hypergalactosemia. To date almost all reported patients were from East Asia and only few cases from Caucasian origin have been described. We report the first Bulgarian case of NICCD. Mutation screening of the SLC25A13 gene revealed the compound heterozygous mutations c.1081C>T (p.R361*) and c.74C>A (p. A25E) which confirmed the diagnosis of NICCD. The nonsense mutation c.1081C>T (p.R361*) is novel.
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