Acute fatty liver of pregnancy associated with fetal mitochondrial trifunctional protein deficiency

J Obstet Gynaecol Res. 2015 May;41(5):799-802. doi: 10.1111/jog.12609. Epub 2014 Nov 25.

Abstract

Acute fatty liver of pregnancy (AFLP) is a devastating disorder of the maternal liver in the third trimester. Recent studies have demonstrated an association between AFLP and fetal fatty acid oxidation disorders. Here, we report a case of AFLP caused by fetal mitochondrial trifunctional protein (TFP) deficiency. A 21-year-old parous woman presented with nausea, genital bleeding and abdominal pain at 33 weeks of gestation. Laboratory data revealed hepatic failure and disseminated intravascular coagulopathy. The patient underwent emergency cesarean section and was diagnosed with AFLP from the clinical characteristics. She was successfully treated with frequent plasma exchange. The newborn presented severe heart failure and died on the 39th day after birth. Tandem mass spectrometry indicated long-chain fatty acid oxidation disorder. Gene analysis demonstrated homozygous mutation in exon 13 of HADHB, the gene responsible for mitochondrial TFP deficiency. The parents carried a heterozygous mutation at the same location in HADHB.

Keywords: acute fatty liver of pregnancy; coagulopathy; fatty acid oxidation disorder; gene analysis; mitochondrial trifunctional protein deficiency.

Publication types

  • Case Reports

MeSH terms

  • Cardiomyopathies / complications*
  • Cardiomyopathies / genetics
  • Fatal Outcome
  • Fatty Liver / etiology*
  • Fatty Liver / therapy
  • Female
  • Heart Failure / complications*
  • Heart Failure / genetics
  • Humans
  • Infant, Newborn
  • Lipid Metabolism, Inborn Errors / complications*
  • Lipid Metabolism, Inborn Errors / genetics
  • Mitochondrial Myopathies / complications*
  • Mitochondrial Myopathies / genetics
  • Mitochondrial Trifunctional Protein / deficiency*
  • Mitochondrial Trifunctional Protein / genetics
  • Mitochondrial Trifunctional Protein, beta Subunit / genetics
  • Mutation
  • Nervous System Diseases / complications*
  • Nervous System Diseases / genetics
  • Plasma Exchange
  • Pregnancy
  • Pregnancy Complications / etiology*
  • Pregnancy Complications / therapy
  • Rhabdomyolysis / complications*
  • Rhabdomyolysis / genetics
  • Young Adult

Substances

  • HADHB protein, human
  • Mitochondrial Trifunctional Protein
  • Mitochondrial Trifunctional Protein, beta Subunit

Supplementary concepts

  • Acute fatty liver of pregnancy
  • Trifunctional Protein Deficiency With Myopathy And Neuropathy