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A newborn has an elevated phenylalanine on newborn screening. Without treatment, affected children develop intellectual disability, seizures, and musty body odor.

AR; PAH (phenylalanine hydroxylase)

  • Untreated: Severe ID, seizures, eczema, light pigmentation, musty odor
  • Newborn screening: Universal (on RUSP)
  • Maternal PKU: Microcephaly, CHD, ID in offspring if mother's Phe uncontrolled
  • Newborn screening (universal, on RUSP): elevated phenylalanine and elevated Phe/tyrosine ratio
  • Confirmatory plasma amino acids: elevated phenylalanine with normal/low tyrosine
  • Exclude BH4 (tetrahydrobiopterin) cofactor defects (pterin analysis, dihydropteridine reductase assay), which present similarly but need different treatment
  • PAH molecular testing confirms classic PKU and predicts BH4 responsiveness
  • Phenylalanine-restricted diet (lifelong), sapropterin (BH4 cofactor) for responsive patients, pegvaliase

"Mousy/musty odor": the characteristic body odor of untreated PKU. Phenylalanine accumulates because it cannot be converted to tyrosine.

Avoid aspartame: aspartame (artificial sweetener) is metabolized to phenylalanine. PKU patients must also avoid high-protein foods.

Tyrosine becomes essential: since PAH (phenylalanine hydroxylase) normally converts Phe to Tyr, tyrosine becomes an essential amino acid in PKU. Decreased tyrosine explains the light skin/hair (less melanin). BH4 (tetrahydrobiopterin) is the cofactor; sapropterin is synthetic BH4.