Rare case of primary carnitine deficiency presenting as acute liver failure

BMJ Case Rep. 2022 Jul 19;15(7):e247225. doi: 10.1136/bcr-2021-247225.

Abstract

Systemic primary carnitine deficiency (PCD) is an autosomal recessive disorder caused by mutations in the SLC22A5 gene that encodes carnitine transporter, OCTN2. Transporter deficiency leads to defective fatty acid oxidation. Signs and symptoms ranging from liver injury in children to cardiomyopathy and skeletal myopathy in adults, manifest during periods of stress and fasting. Though acute liver failure is infrequently described, young children presenting as acute liver failure should be screened for fatty acid oxidation defects including PCD by testing plasma for amino acids and further confirmed by genetic sequencing. Early identification and treatment using L-carnitine is lifesaving. Our patient presented as acute liver failure and diagnosis of PCD was confirmed by metabolic screening and genetic sequencing. He responded to the treatment.

Keywords: Genetics; Liver disease; Paediatrics; Paediatrics (drugs and medicines).

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Cardiomyopathies* / diagnosis
  • Cardiomyopathies* / genetics
  • Carnitine / deficiency
  • Carnitine / therapeutic use
  • Child
  • Child, Preschool
  • Fatty Acids
  • Humans
  • Hyperammonemia
  • Liver Failure, Acute* / diagnosis
  • Liver Failure, Acute* / etiology
  • Male
  • Muscular Diseases* / diagnosis
  • Muscular Diseases* / genetics
  • Mutation
  • Organic Cation Transport Proteins / genetics
  • Solute Carrier Family 22 Member 5 / genetics

Substances

  • Fatty Acids
  • Organic Cation Transport Proteins
  • SLC22A5 protein, human
  • Solute Carrier Family 22 Member 5
  • Carnitine

Supplementary concepts

  • Systemic carnitine deficiency