Literature DB >> 27099654

Management of pregnancy in Crigler Najjar syndrome type 2.

Alisha Nitin Chaubal1, Ruchir Patel1, Dhaval Choksi1, Kaivan Shah1, Meghraj Ingle1, Prabha Sawant1.   

Abstract

Crigler Najjar syndrome is associated with indirect hyperbilirubinemia due to a deficiency of enzyme Uridine Di Phospho Glucoronosyl Transferase (UDPGT). Presented here is a case of a female in the first trimester of pregnancy, who was diagnosed to have type 2 Crigler Najjar syndrome. We also discuss the management of this rare disease especially in pregnancy. Unconjugated bilirubin can cross the placental barrier causing neurological damage in the newborn. Patient was carefully monitored during pregnancy and treatment with phenobarbitone in low doses was adjusted such that the serum bilirubin levels were below 10 mg/dL. Crigler Najjar syndrome being rare needs to be diagnosed early in pregnancy to avoid adverse fetal outcomes. Phenobarbitone being an inducer of enzyme UDPGT is used as the first line of treatment and is not teratogenic in the low doses used. Treatment protocol followed was on the basis of previous reported cases and successful perinatal outcome was achieved.

Entities:  

Keywords:  Crigler Najjar type 2; Folic acid; Kernicterus; Phenobarbitone; Pregnancy

Year:  2016        PMID: 27099654      PMCID: PMC4832095          DOI: 10.4254/wjh.v8.i11.530

Source DB:  PubMed          Journal:  World J Hepatol


  10 in total

1.  Use of phenytoin, phenobarbital, or diazepam during pregnancy and risk of congenital abnormalities: a case-time-control study.

Authors:  Dorte Kjaer; Erzsébet Horvath-Puhó; Jakob Christensen; Mogens Vestergaard; Andrew E Czeizel; Henrik Toft Sørensen; Jørn Olsen
Journal:  Pharmacoepidemiol Drug Saf       Date:  2007-02       Impact factor: 2.890

2.  Prenatal exposure to conjugated bilirubin.

Authors:  Francesco Raimondi; Letizia Capasso; Fiorella Migliaro; Antonia Romano; Roberto Paludetto
Journal:  Pediatrics       Date:  2006-11       Impact factor: 7.124

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Authors:  J F Smith; J M Baker
Journal:  Obstet Gynecol       Date:  1994-10       Impact factor: 7.661

4.  Pregnancy in Crigler-Najjar syndrome. Case report.

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Journal:  Br J Obstet Gynaecol       Date:  1991-12

5.  Recommendations for pregnancies in patients with crigler-najjar syndrome.

Authors:  J H Paul Wilson; Maarten Sinaasappel; Fred K Lotgering; Janneke G Langendonk
Journal:  JIMD Rep       Date:  2012-04-22

6.  Successful photo-and phenobarbital therapy during pregnancy in a woman with Crigler-Najjar syndrome type II.

Authors:  Andreas Holstein; Armin Plaschke; Peter Lohse; Eick-Hartwig Egberts
Journal:  Scand J Gastroenterol       Date:  2005-09       Impact factor: 2.423

Review 7.  Genetic lesions of bilirubin uridine-diphosphoglucuronate glucuronosyltransferase (UGT1A1) causing Crigler-Najjar and Gilbert syndromes: correlation of genotype to phenotype.

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Journal:  Hum Mutat       Date:  2000-10       Impact factor: 4.878

Review 8.  Definition of the clinical spectrum of kernicterus and bilirubin-induced neurologic dysfunction (BIND).

Authors:  Steven M Shapiro
Journal:  J Perinatol       Date:  2005-01       Impact factor: 2.521

9.  Evidence for carrier-mediated transport of unconjugated bilirubin across plasma membrane vesicles from human placental trophoblast.

Authors:  M A Serrano; J E Bayón; L Pascolo; C Tiribelli; J D Ostrow; J Gonzalez-Gallego; J J G Marin
Journal:  Placenta       Date:  2002-08       Impact factor: 3.481

Review 10.  Pregnancy outcome in maternal Crigler-Najjar syndrome type II: a case report and systematic review of the literature.

Authors:  V Passuello; A G Puhl; S Wirth; E Steiner; C Skala; H Koelbl; N Kohlschmidt
Journal:  Fetal Diagn Ther       Date:  2009-09-11       Impact factor: 2.587

  10 in total

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