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A plasma ammonia above the age-specific upper limit in a symptomatic patient (roughly > 100 µmol/L in a neonate, > 80 µmol/L beyond infancy, with anything > 150 µmol/L treated as a metabolic emergency). The clinical picture is encephalopathy out of proportion to the rest of the labs: lethargy, poor feeding, vomiting, tachypnea, progressive obtundation, seizures, cerebral edema. Ammonia is directly neurotoxic; every hour the brain sits at > 200 µmol/L compounds long-term injury, and dialysis is on the table early.

The cardinal question in front of a hyperammonemic patient is whether acidosis is present.

  1. Hyperammonemia WITHOUT acidosis (often respiratory alkalosis from CNS hyperventilation) → urea cycle defect until proven otherwise.
  2. Hyperammonemia WITH high anion-gap acidosis and ketosis → organic acidemia (propionic acidemia, methylmalonic acidemia, isovaleric acidemia).

Once a UCD is suspected, the next two labs decide the gene: plasma citrulline (proximal vs distal block) and urine orotic acid (cytosolic vs mitochondrial enzyme).

Urea cycle defects (algorithm by citrulline and orotic acid)

Plasma citrullineUrine orotic acidDisorderInheritance
LowLowNAGS deficiencyAR
LowLowCPS1 deficiencyAR
LowHighOTC deficiencyX-linked
Very highHighASS1 (citrullinemia type 1)AR
Moderately high (+ ASA in urine)HighASL deficiency (argininosuccinic aciduria)AR
Normal-low (high arginine)VariableARG1 (arginase) deficiencyAR
High ornithine + homocitrullinuriaHighHHH syndrome (SLC25A15)AR

The exam favorite is OTC deficiency: an X-linked, partially-penetrant cause that classically presents as a previously well boy or a manifesting female carrier with episodic hyperammonemia after a protein load, illness, or postpartum catabolism. Low citrulline plus high orotic acid is the splitting branch off CPS1/NAGS.

Organic acidemias (ammonia AND acidosis AND ketosis)

Propionic, methylmalonic, and isovaleric acidemias inhibit N-acetylglutamate synthase secondarily, so ammonia rises alongside the dominant high anion-gap acidosis. Urine organic acids and plasma acylcarnitine profile crack the case (C3 in PA/MMA, C5 in IVA).

Other causes worth a glance

  • Liver failure of any etiology (galactosemia, tyrosinemia, neonatal hemochromatosis).
  • Portosystemic shunt (Abernethy malformation) presenting in older children.
  • Valproate-induced (often unmasks an underlying OTC carrier).
  • Pyruvate carboxylase deficiency, lysinuric protein intolerance (rare).
  • No acidosis, often respiratory alkalosis → urea cycle defect.
  • High anion-gap acidosis with ketosis → organic acidemia.
  • Low citrulline + high urine orotic acidOTC (X-linked; manifesting female carriers exist).
  • Massively elevated citrullineASS1 (citrullinemia type 1).
  • Argininosuccinic acid detectable in urineASL.
  • Previously well child on the third postpartum day with progressive lethargy → late-onset OTC (or another UCD) until disproven.
  • Valproate-precipitated coma → think undiagnosed OTC carrier.

In a patient with ammonia > 150 µmol/L, treatment and diagnosis run in parallel. Don't wait on labs to escalate.

  1. Repeat ammonia free-flowing, on ice, run STAT. Tourniquet artifact and delayed processing falsely elevate.
  2. Venous blood gas, glucose, lactate, electrolytes (anion gap), ketones: classify the metabolic phenotype.
  3. Plasma amino acids (citrulline is the branch point), plasma acylcarnitine profile (PA/MMA/IVA signatures), urine organic acids, urine orotic acid.
  4. Liver function (rule out hepatic failure as the cause).
  5. Empiric treatment while results return: stop protein intake, start D10 IV at 1.5× maintenance to suppress catabolism, give IV nitrogen-scavengers (sodium benzoate + sodium phenylacetate, marketed as Ammonul), IV L-arginine (provides substrate for the urea cycle distal to CPS1).
  6. Hemodialysis (preferred over CRRT for speed) for ammonia > 500 µmol/L, rising despite scavengers, or coma. Peritoneal dialysis and exchange transfusion are slower and last-resort.
  7. Targeted gene sequencing once the biochemical phenotype is in hand; broad metabolic panel or exome if unclear.
  • No acidosis + high ammonia = UCD. The single most useful initial sorting rule. Respiratory alkalosis (from central hyperventilation) is the tell.
  • Citrulline is the gene-localizer. Low + high orotic = OTC. Very high citrulline = ASS1. Argininosuccinic acid = ASL.
  • OTC is X-linked but female carriers can present, sometimes triggered by pregnancy, surgery, or valproate. Don't reject OTC in a girl.
  • Ammonia > 500 = dialysis. Hours of hyperammonemic coma map onto IQ points lost. Call nephrology before the labs are back.
  • Newborn screening catches most organic acidemias before the first crisis; a child in crisis is often pre-screen or false-negative. Re-check the screen.