Successful treatment of a patient with ethylmalonic encephalopathy by intravenous N-acetylcysteine

Metab Brain Dis. 2017 Apr;32(2):293-296. doi: 10.1007/s11011-016-9928-5. Epub 2016 Nov 9.

Abstract

Ethylmalonic encephalopathy (EE) is an autosomal recessive devastating metabolic disorder affecting the brain, gastrointestinal tract, peripheral vessels and rarely the other vascular organs. We report a 10-month-old girl who presented as a meningococcemia clinic but later diagnosed ethylmalonic encephalopathy. Molecular analyses revealed a homozygous c.554 T > G; p. L185R mutation in ETHE1 gene. She was only partially benefited from riboflavine, coenzyme Q10, metronidazole, N-acetylcysteine and symptomatic treatment and discharged from hospital with the sequela of oxygene dependance and developmental delay. We observed N-acetylcysteine 100 mg/kg/day intravenous infusion theraphy may be the most important drug especially in comatous EE patients.

Keywords: Acrocyanosis; Developmental delay; ETHE1; Ethylmalonic encephalopathy; Meningococcemia; Recurrent petechia.

Publication types

  • Case Reports

MeSH terms

  • Acetylcysteine / administration & dosage
  • Acetylcysteine / therapeutic use*
  • Brain Diseases, Metabolic, Inborn / diagnostic imaging
  • Brain Diseases, Metabolic, Inborn / drug therapy*
  • Brain Diseases, Metabolic, Inborn / metabolism
  • Coma / drug therapy
  • Coma / etiology
  • Female
  • Humans
  • Infant
  • Infusions, Intravenous
  • Magnetic Resonance Imaging
  • Malonates
  • Mitochondrial Proteins / genetics
  • Nucleocytoplasmic Transport Proteins / genetics
  • Purpura / diagnostic imaging
  • Purpura / drug therapy*
  • Purpura / metabolism

Substances

  • ETHE1 protein, human
  • Malonates
  • Mitochondrial Proteins
  • Nucleocytoplasmic Transport Proteins
  • Acetylcysteine

Supplementary concepts

  • Ethylmalonic encephalopathy