Prenatal molecular diagnosis of inherited cholestatic diseases

J Pediatr Gastroenterol Nutr. 2007 Apr;44(4):453-8. doi: 10.1097/MPG.0b013e318036a569.

Abstract

Objectives: Progressive familial intrahepatic cholestasis (PFIC) and to a lesser extent, Alagille syndrome, often lead to end-stage liver disease during childhood. We report our experience of DNA-based prenatal diagnosis of PFIC1-3 and Alagille syndrome.

Patients and methods: Four molecular antenatal diagnoses were performed in 3 PFIC families and 17 in 11 Alagille syndrome families. DNA was isolated from chorionic villus or cultured amniocyte samples from women, without pregnancy complications.

Results: All four foetuses with a family history of PFIC1, 2, or 3 were heterozygous for an ATP8B1, ABCB11, or ABCB4 mutation and pregnancies were continued. Three of the infants were healthy after birth, and 1 premature infant, who had an ABCB4 mutation, experienced transient neonatal cholestasis. Among the families with a history of de novo JAG1 mutation, none of the foetuses was mutated, versus 40% of those with a history of familial mutation. Of 4 pregnant women with a JAG1-mutated foetus, 3 cut short their pregnancy and 1 gave birth to a child with overt Alagille syndrome.

Conclusions: Molecular antenatal diagnosis of PFIC1-3 and Alagille syndrome is reliable because clinical outcome after birth corresponded to molecular foetal data.

MeSH terms

  • Alagille Syndrome / diagnosis
  • Alagille Syndrome / genetics
  • Cholestasis, Intrahepatic / diagnosis*
  • Cholestasis, Intrahepatic / genetics*
  • Chorionic Villi Sampling
  • DNA / analysis
  • Female
  • Genetic Counseling
  • Humans
  • Mutation
  • Pregnancy

Substances

  • DNA