Definition: inherited genetic disorder characterized by impaired purine salvage pathway, resulting in an overproduction of uric acid

Epidemiology


Etiology


Pathophysiology

Purine Salvage Pathway

  • Goal: Recycle free purine bases to reform nucleotides, conserving energy.
  • Key Enzymes:
    • HGPRT (Hypoxanthine-guanine phosphoribosyltransferase):
      • Converts Hypoxanthine → IMP.
      • Converts Guanine → GMP.
    • APRT (Adenine phosphoribosyltransferase):
      • Converts Adenine → AMP.
  • Lesch-Nyhan SyndromeX-linked recessive deficiency of HGPRT.
    • Patho: Inability to salvage hypoxanthine and guanine leads to their degradation, causing excess uric acid production. Also leads to ↑ de novo purine synthesis due to ↑ PRPP levels.
    • Presentation (HGPRT):
      • Hyperuricemia (gout, “orange sand” [sodium urate crystals] in diaper).
      • Gout.
      • Pissed off (aggression, self-mutilation).
      • Retardation (intellectual disability).
      • Tonia (dystonia).

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Clinical features

  • Usually asymptomatic for the first 6 months of life
  • Orange sand-like sodium urate crystals can be found in the diapers of infants with hyperuricemia.
  • Developmental delay and cognitive impairment
  • Pyramidal and extrapyramidal symptoms (e.g., dystonia, spasticity)
  • Gouty arthritis, urate nephropathy
    • Due to the accumulation of uric acid in peripheral tissues
  • Aggression, self-injurious behavior
    • E.g., finger chewing, lip biting, headbanging
  • Renal failure

Diagnostics

  • Hyperuricemia
  • ↓ HGPRT activity
  • ↑ Phosphoribosyl pyrophosphate amidotransferase (converts PRPP to GMP and AMP)
  • Macrocytosis (megaloblastic anemia may occur)

Treatment

  • Reduce uric acid levels:
    • Allopurinol (first-line)
    • Febuxostat (second-line)
    • Low-purine diet