Department of Neonatology, Children's Hospital of Fudan University, Shanghai 201102, China.
Chin Med J (Engl). 2011 Dec;124(23):4109-11.
We present a case of severe persisting unconjugated hyperbilirubinemia in a Uigur infant boy, eventually diagnosed as Crigler-Najjar syndrome type I. DNA analysis of his blood of the UGT1A1 gene sequence demonstrated that he was homozygous for an insertion mutation causing a change of the coding exons with a frame-shift, resulting in the substitution of 27 abnormal amino acid residues in his hepatic bilirubin uridine diphosphoglucuronyl transferase enzyme. Both of his parents were heterozygous for the same mutation. A novel frame-shifting mutation of the UGT1A1 gene was found, confirming the diagnosis of Crigler-Najjar syndrome type I for this patient.
我们报告一例维吾尔族婴儿男孩严重持续性未结合高胆红素血症,最终诊断为克里格勒-纳贾尔综合征 I 型。对其血液 UGT1A1 基因序列的 DNA 分析表明,他是导致编码外显子移码突变的纯合子,导致其肝胆红素尿苷二磷酸葡萄糖醛酸基转移酶中替换 27 个异常氨基酸残基。他的父母均为同一突变的杂合子。发现 UGT1A1 基因的一种新的移码突变,从而确诊该患者为克里格勒-纳贾尔综合征 I 型。