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Arginase deficiency (argininemia)

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A child presents with progressive spastic diplegia and intellectual disability. Unlike other urea cycle disorders, hyperammonemia is mild or absent. Plasma arginine is markedly elevated.

AR; ARG1 (arginase 1)

  • Progressive spastic paraparesis (resembles cerebral palsy)
  • Intellectual disability
  • Seizures
  • Milder/absent hyperammonemia (unlike other UCDs)
  • Markedly elevated plasma arginine
  • Growth restriction
  • Presents later than other UCDs (1-3 years)
  • On RUSP
  • Newborn screening (RUSP) flags elevated arginine on tandem mass spectrometry
  • Plasma amino acids: markedly elevated arginine (the diagnostic biochemistry, distinct from other urea cycle disorders)
  • Reduced/absent erythrocyte arginase enzyme activity
  • Confirmatory ARG1 molecular testing
  • Protein restriction (especially arginine), nitrogen scavengers if needed

Arginase is the last step of the urea cycle: the first 3 urea cycle enzymes are intramitochondrial (NAGS, CPSI, OTC) and the last 3 are cytosolic (ASS1, ASL, Arginase). Arginase converts arginine to ornithine + urea.

"The spastic UCD": arginase deficiency uniquely presents with progressive spastic diplegia (resembles cerebral palsy), unlike other UCDs which present with acute hyperammonemic crises. Hyperammonemia is mild or absent.

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