Crigler - Najjar综合征-儿童黄疸的罕见病例

Bodhrun Naher, M. Mazumder, S. Ghosal, A. R. Rahman, A. Karim
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摘要

Crigler-Najjar综合征(Crigler-Najjar syndrome, CNS)于1952年在美国马里兰州首次被Crigler JF和Najjar va描述为先天性家族性非溶血性黄疸伴核黄疸。CNS是一种罕见的遗传性疾病,以胆红素代谢异常为特征,以非共轭胆红素持续升高为明显特征。在生命的最初几天,该综合征的临床表现为强烈的非共轭高胆红素血症,没有溶血的证据。它包括两种类型,I型和II型。Crigler-Najjar综合征多为常染色体隐性遗传病,但CNSⅱ的遗传也可能发生变异。2主要发病机制是由于尿苷5′-二磷酸-葡萄糖醛酸转移酶(UGT)完全或部分缺乏导致胆红素偶联缺陷。这种酶是结合和进一步从体内排出胆红素所必需的。在I型中枢神经系统中,酶活性完全缺失,而在II型中枢神经系统中,酶部分缺失。因此,I型是更严重的形式,通常在1-2岁时与核黄疸致命。3-5型病情较轻,预后较好。CN II型患者黄疸较少,神经功能受损较少,对苯巴比妥治疗反应良好(血清胆红素水平至少降低25%)男性和女性都同样受到中枢神经系统的影响。在一般人群中发病率约为75 - 100万分之一在此我们报告这样一个罕见的病例。《北方国际医学院学报》2020年7月第12卷第1期,第515-517页
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Crigler Najjar Syndrome - A Rare case of Jaundice in Children
Crigler-Najjar syndrome (CNS) was first described in 1952 in Maryland, USA as congenital familial non-hemolytic jaundice with kernicterus by Crigler JF and Najjar VA.1 CNS is a rare genetic disorder characterized by abnormalities in bilirubin metabolism and evident by persistent increase of unconjugated bilirubin. During the first days of life, the syndrome clinically manifests as intense unconjugated hyperbilirubinemia without evidence of hemolysis. It consists of two types, type I and type II. Crigler-Najjar Syndrome is mostly autosomal recessive disorder, but variation may occur in the inheritance of CNS II.2 The key pathogenesis is defect in bilirubin conjugation due to complete or partial deficiency of uridine 5'-diphosphate-glucuronosyl transferase (UGT). This enzyme is required for the conjugation and further excretion of bilirubin from the body. In type I CNS the enzyme activity is completely absent and in type II there is partial absence of the enzyme. Therefore, Type I is more severe form and usually fatal with kernicterus at the age of 1-2 years.3-5 TypeII is less severe and has better prognosis. Patients with CN type II suffer from less jaundice, less neurological impairment, and show a fair response to phenobarbitone therapy (serum bilirubin levels decrease by at least 25%).6 Both males and females are equally affected by CNS. The incidence is approximately 1 in 750,000-1,000,000 in the general population.7 Here we report such a rare case. Northern International Medical College Journal Vol. 12 No.1 July 2020, Page 515-517
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