[Clinical features and gene mutations in a patient with multiple aeyl-CoA dehydrogenase deficiency with severe fatty liver]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2016 Apr;33(2):191-4. doi: 10.3760/cma.j.issn.1003-9406.2016.02.014.
[Article in Chinese]

Abstract

Objective: To analyze the clinical features and gene mutations in an adolescent patient affected with late-onset multiple aeyl-CoA dehydrogenase deficiency (MADD) with severe fatty liver.

Methods: Potential mutations of the ETFDH gene were detected with polymerase chain reaction (PCR) and DNA sequencing.

Results: The 13-year-and-10-month girl has presented with weakness without any other special manifestation. Laboratory tests demonstrated an elevation of myocardial enzyme levels, total cholesterol, lactic acid and abnormal serum free fatty acids. H magnetic resonance spectroscopy revealed severe fatty liver. An increase in multiple plasma acyl-carnitines was detected by gas chromatography/mass spectrometry and isobutyrylglycine in urine by screening with tandem mass spectrometry. Genetic analysis demonstrated 2 heterozygous missense mutations c.250G>A (p.Ala84Thr) and c.353G>T (p.Cys118Phe) in the ETFDH gene. The diagnosis of MADD was confirmed. The patient was given large dose of vitamin B2, which resulted in rapid clinical and biochemical improvement.

Conclusion: A common mutation c.250G>A and a novel mutation c.353G>T in the ETFDH gene were identified in the patient. The pathogenic role of c.353G>T (p.Cys118Phe) deserves further study. Early diagnosis of MADD and appropriate therapy is crucial for the prognosis.

Publication types

  • English Abstract
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Base Sequence
  • Electron-Transferring Flavoproteins / genetics*
  • Fatty Acids, Nonesterified / blood
  • Fatty Liver / blood
  • Fatty Liver / enzymology
  • Fatty Liver / genetics*
  • Female
  • Humans
  • Infant
  • Iron-Sulfur Proteins / genetics*
  • Male
  • Molecular Sequence Data
  • Multiple Acyl Coenzyme A Dehydrogenase Deficiency / blood
  • Multiple Acyl Coenzyme A Dehydrogenase Deficiency / enzymology
  • Multiple Acyl Coenzyme A Dehydrogenase Deficiency / genetics*
  • Mutation
  • Oxidoreductases Acting on CH-NH Group Donors / genetics*
  • Pedigree

Substances

  • Electron-Transferring Flavoproteins
  • Fatty Acids, Nonesterified
  • Iron-Sulfur Proteins
  • Oxidoreductases Acting on CH-NH Group Donors
  • electron-transferring-flavoprotein dehydrogenase