Singh Sukanya, Tayade Surekha, Makhija Nidhi, Patel Drashti, Singh Akanksha
Department of Obstetrics and Gynaecology, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences, Wardha, IND.
Department of Obstetrics and Gynaecology, Institute of Post Graduate Medical Education, Research and Seth Sukhlal Karnani Memorial Hospital, Kolkata, IND.
Cureus. 2024 Apr 26;16(4):e59075. doi: 10.7759/cureus.59075. eCollection 2024 Apr.
Crigler-Najjar syndrome (CNS) is a genetic syndrome that results in increased levels of unconjugated bilirubin due to less or completely nonfunctional enzyme, uridine diphosphoglucoronyltransferase (UDPGT) in hepatocytes. When bilirubin metabolism is compromised, hyperbilirubinemia is caused, which results in increased levels of unconjugated and conjugated bilirubin in the bloodstream. CNS is an autosomal recessive disorder, usually noticeable as people get older. This disorder is divided into two types: CNS type I and CNS type II, which are caused by homozygous or compound heterozygous mutations in the UDP glucuronosyltransferase family 1 member A1 (UGT1A1) gene. The disorder affects all races and genders equally, with a prevalence of one per million births. CNS type I is more severe and has almost undetectable UDPGT expression activity, and affected individuals die before one year of age. Consanguineous marriages are a major risk factor as CNS is inherited in an autosomal recessive manner. Being rare, maternal CNS type II is yet to be completely understood in terms of its impact on the mother, her pregnancy, and the infant. We aim to present a case of a pregnant female with CNS type II and its clinical course. She was monitored closely during her pregnancy. The treatment protocol was followed as per previously reported cases and was managed on low, non-teratogenic doses of phenobarbitone. A successful outcome with the birth of a healthy infant having normal neurological development till six months follow-up was observed.
克里格勒 - 纳贾尔综合征(CNS)是一种遗传性综合征,由于肝细胞中尿苷二磷酸葡萄糖醛酸基转移酶(UDPGT)减少或完全无功能,导致未结合胆红素水平升高。当胆红素代谢受损时,会引起高胆红素血症,导致血液中未结合胆红素和结合胆红素水平升高。CNS是一种常染色体隐性疾病,通常随着年龄增长而变得明显。这种疾病分为两种类型:CNS I型和CNS II型,由UDP葡萄糖醛酸基转移酶家族1成员A1(UGT1A1)基因的纯合或复合杂合突变引起。该疾病对所有种族和性别影响相同,每百万例出生中的患病率为1例。CNS I型更为严重,UDPGT表达活性几乎无法检测到,受影响个体在一岁前死亡。近亲结婚是一个主要风险因素,因为CNS是以常染色体隐性方式遗传的。由于罕见,母亲的CNS II型对母亲、其妊娠和婴儿的影响尚未完全了解。我们旨在介绍一例患有CNS II型的孕妇及其临床过程。她在怀孕期间受到密切监测。按照先前报道的病例遵循治疗方案,并采用低剂量、无致畸性的苯巴比妥进行治疗。观察到成功分娩出一名健康婴儿,直至六个月随访时神经发育正常。