Molecular prenatal diagnosis in families with fetal mitochondrial trifunctional protein mutations

J Pediatr. 2001 Mar;138(3):396-9. doi: 10.1067/mpd.2001.111503.

Abstract

Objectives: To evaluate the feasibility of molecular prenatal diagnosis in families with mitochondrial trifunctional protein (TFP) mutations and prospectively study the effects of fetal genotype on pregnancy outcome. TFP catalyzes the last 3 steps in mitochondrial long-chain fatty acid oxidation.

Study design: We performed molecular prenatal diagnosis in 9 pregnancies, 8 in 6 families with isolated long-chain 3-hydroxyacyl-coenzyme A dehydrogenase (LCHAD) deficiency and one in a family with complete TFP deficiency. Analyses were performed on chorionic villous samples in 7 pregnancies and on amniocytes in 2.

Results: Molecular prenatal diagnosis successfully identified the fetal genotype in all 9 pregnancies. Two fetuses were affected, and both pregnancies were terminated by family decision. Two other fetuses had normal genotype and 5 others were heterozygotes. These 7 pregnancies were uncomplicated, and all the offspring are alive and apparently healthy. Genotypes of the aborted fetuses and neonates were confirmed by molecular analysis and enzymatic assays.

Conclusions: Molecular prenatal diagnosis is possible and valid in guiding management of pregnancies in families with known TFP defects. Women heterozygous for TFP alpha-subunit mutations who carry fetuses with wild-type or heterozygous genotypes have uncomplicated pregnancies.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Amniocentesis*
  • Chorionic Villi Sampling*
  • Fatty Liver / etiology
  • Female
  • Genotype
  • HELLP Syndrome / etiology
  • Humans
  • Lipid Metabolism, Inborn Errors / diagnosis*
  • Lipid Metabolism, Inborn Errors / genetics
  • Mitochondrial Trifunctional Protein
  • Multienzyme Complexes / deficiency*
  • Multienzyme Complexes / genetics
  • Palmitoyl-CoA Hydrolase / deficiency*
  • Palmitoyl-CoA Hydrolase / genetics
  • Pregnancy
  • Pregnancy Complications / etiology
  • Pregnancy Outcome

Substances

  • Multienzyme Complexes
  • Mitochondrial Trifunctional Protein
  • Palmitoyl-CoA Hydrolase