Overview

Definition:
-Hyperammonemia refers to an abnormally high concentration of ammonia in the blood, exceeding the normal physiological range
-It is a critical biochemical abnormality often indicative of an underlying inborn error of metabolism (IEM), most commonly a disorder of the urea cycle, which impairs the body's ability to detoxify ammonia
-This can lead to severe neurological dysfunction and is a medical emergency.
Epidemiology:
-The incidence of hyperammonemia varies depending on the underlying cause
-Urea cycle disorders (UCDs) affect approximately 1 in 30,000 live births
-Ornithine transcarbamylase (OTC) deficiency is the most common UCD, accounting for about 50% of cases
-Other causes include organic acidemias (e.g., propionic acidemia, methylmalonic acidemia), fatty acid oxidation defects, and certain inherited transport defects
-Presentation can occur in neonates, infants, or older children, with neonatal onset often being more severe.
Clinical Significance:
-Elevated ammonia levels are neurotoxic, leading to encephalopathy, coma, and potentially irreversible brain damage or death if not managed promptly
-Prompt recognition and intervention are crucial for improving neurological outcomes
-This condition is a high-yield topic for DNB and NEET SS examinations due to its emergent nature and the need for rapid, evidence-based management.

Clinical Presentation

Symptoms:
-Neonatal presentation: Lethargy
-Poor feeding
-Vomiting
-Irritability
-Seizures
-Jaundice
-Hypotonia
-Respiratory distress
-Older infants/children: Recurrent vomiting
-Anorexia
-Lethargy
-Developmental delay or regression
-Behavioral changes
-Ataxia
-Seizures
-Coma.
Signs:
-Neurological: Altered mental status ranging from lethargy to deep coma
-Seizures (generalized, focal, or myoclonic)
-Hypertonia or hypotonia
-Irritability
-Signs of increased intracranial pressure (bulging fontanelle, papilledema - late)
-Hepatic: Hepatomegaly may be present in some underlying conditions
-Other: Poor weight gain, recurrent infections, developmental delay.
Diagnostic Criteria:
-No single definitive diagnostic criterion exists beyond elevated serum ammonia levels
-Diagnosis relies on a combination of clinical suspicion, laboratory findings (elevated ammonia, specific IEM markers), and confirmation with genetic testing
-Levels > 150-200 µmol/L in neonates or > 100-150 µmol/L in older children are generally considered significantly elevated and warrant urgent investigation.

Diagnostic Approach

History Taking:
-Detailed birth history (gestational age, complications)
-Feeding history (vomiting, poor intake)
-Developmental milestones
-Previous unexplained illnesses or episodes of lethargy/seizures
-Family history of unexplained infant deaths, neurological disorders, or known IEMs
-Maternal history of consanguinity
-Dietary history, especially protein intake
-Red Flags: Acute onset of neurological symptoms with metabolic derangement, unexplained lethargy, poor feeding, vomiting, seizures, especially in the neonatal period or in a previously well child.
Physical Examination:
-Complete neurological examination: Assess level of consciousness (AVPU, GCS)
-Evaluate for focal neurological deficits, seizures, tone abnormalities (hyper/hypotonia)
-Assess for signs of increased intracranial pressure
-Thorough abdominal examination for hepatomegaly or masses
-Assess hydration status and vital signs, including temperature.
Investigations:
-Immediate: Serum ammonia (on ice, arterial or venous, no tourniquet, prompt transport)
-Blood glucose
-Electrolytes, BUN, creatinine
-Liver function tests (ALT, AST, bilirubin, albumin, PT/INR)
-Arterial blood gas (ABG) to assess for metabolic acidosis
-Urine analysis and screen for ketones
-Consider: Plasma amino acids (elevated glutamine is common)
-Urine organic acids
-Serum lactate
-Blood acylcarnitines
-Ammonia level recheck after initial management to assess response
-Diagnostic imaging: Cranial ultrasound or CT/MRI to rule out structural brain lesions or cerebral edema, especially if seizures are present or neurological signs are focal.
Differential Diagnosis:
-Sepsis (especially gram-negative)
-Hypoglycemia
-Hypoxic-ischemic encephalopathy
-Intracranial hemorrhage
-Inborn errors of metabolism (UCDs, organic acidemias, fatty acid oxidation defects, amino acid disorders)
-Portosystemic shunting (e.g., congenital hepatic shunts)
-Reye syndrome (historically associated with aspirin use in viral illness)
-Certain drug intoxications.

Management

Initial Management:
-Immediate reduction of ammonia production and promotion of ammonia excretion
-Stop protein intake: Discontinue all oral feeding and administer intravenous fluids with dextrose (10-15%) to provide adequate non-protein calories (120-150 mg/kg/min or 8-10 mg/kg/hr) to prevent catabolism
-Intravenous fluids to maintain hydration and urine output
-Correct any metabolic derangements (e.g., hypoglycemia, electrolyte imbalances)
-Hemodynamic stabilization if shock is present.
Medical Management:
-Specific ammonia-lowering agents: Sodium benzoate and sodium phenylacetate (Ammonul®, Buphenyl®/Ravicti®) - these provide alternative pathways for nitrogen excretion
-Dosing is critical and weight-based
-For example, the standard initial dose for the combination is 250 mg/kg/day for sodium benzoate and 250 mg/kg/day for sodium phenylacetate, divided into 4-6 doses
-Continuous infusion may be used in severe cases
-Glycerol phenylbutyrate (Ravicti®) is another option, given orally or via NG tube, which is converted to phenylacetate
-Dialysis (hemodialysis or peritoneal dialysis): Indicated for severe hyperammonemia (ammonia > 300-500 µmol/L), poor response to medical management, or significant neurological deterioration
-Hemodialysis is the most effective method for rapid ammonia reduction
-Anticonvulsants: For seizures, use benzodiazepines (e.g., lorazepam) followed by other agents if necessary (e.g., levetiracetam, phenobarbital), avoiding valproic acid, which can be ketogenic.
Surgical Management:
-Liver transplantation: Considered for severe, refractory UCDs or when medical management fails to prevent irreversible neurological damage
-This is a definitive treatment but is reserved for specific cases and usually after initial stabilization and diagnosis.
Supportive Care:
-Close monitoring: Frequent monitoring of ammonia levels (every 4-6 hours initially), electrolytes, glucose, and neurological status
-Neurological assessment: Monitor for signs of worsening encephalopathy, seizures, or increased intracranial pressure
-Respiratory support: Ensure adequate oxygenation and ventilation, especially if intubated
-Nutritional support: Once stable, gradual reintroduction of protein with restriction tailored to the specific IEM, guided by a metabolic specialist
-Long-term management: Requires a multidisciplinary team including a metabolic physician, geneticist, dietitian, and neurologist
-Genetic counseling is essential.

Complications

Early Complications:
-Neurological sequelae (developmental delay, intellectual disability, cerebral palsy)
-Seizures refractory to treatment
-Cerebral edema and increased intracranial pressure
-Coma
-Death.
Late Complications:
-Long-term neurodevelopmental deficits
-Learning disabilities
-Behavioral problems
-Recurrent episodes of hyperammonemia if adherence to dietary and medication regimens is poor.
Prevention Strategies:
-Newborn screening programs can identify some IEMs before symptomatic presentation
-Prompt recognition and aggressive management of suspected IEMs in neonates and infants presenting with metabolic encephalopathy
-Genetic counseling and prenatal diagnosis for families with known UCDs
-Strict adherence to dietary restrictions and medication regimens in diagnosed patients.

Prognosis

Factors Affecting Prognosis:
-Severity of hyperammonemia at presentation (peak ammonia level)
-Age at onset (neonatal onset generally has a worse prognosis)
-Underlying specific IEM and its severity
-Rapidity and effectiveness of initial management
-Presence and severity of neurological damage at diagnosis
-Response to treatment, including dialysis if required
-Adherence to long-term management plan.
Outcomes:
-With prompt and aggressive management, some individuals can survive and have a reasonable neurodevelopmental outcome, especially if diagnosed early and treated effectively
-However, survivors often have some degree of neurocognitive impairment
-Untreated or delayed treatment carries a high mortality rate and severe neurological disability.
Follow Up:
-Lifelong follow-up is essential for patients diagnosed with IEMs causing hyperammonemia
-This includes regular monitoring of ammonia levels, metabolic status, growth, and neurodevelopment
-Dietary adjustments and medication management are crucial
-Periodic evaluation by a metabolic specialist and dietitian is required
-Psychological support for patients and families is important.

Key Points

Exam Focus:
-Always suspect IEM in a neonate or infant with unexplained encephalopathy, lethargy, vomiting, or seizures, especially with metabolic acidosis or hyperammonemia
-Differentiate between acute ammonia toxicity and the underlying IEM
-Understand the rapid management steps: stop protein, give non-protein calories, start ammonia scavengers, and consider dialysis urgently for severe cases.
Clinical Pearls:
-Transport blood for ammonia on ice and without delay
-Recognize that a normal glucose level does not rule out an IEM causing hyperammonemia
-Remember that protein restriction alone is insufficient
-aggressive ammonia-lowering strategies are paramount
-Liver transplantation is a definitive treatment for UCDs but not a first-line emergency management.
Common Mistakes:
-Delaying ammonia measurement or management
-Insufficient non-protein caloric support, leading to continued catabolism
-Inadequate doses of ammonia scavengers or delaying their initiation
-Misattributing symptoms to common conditions like sepsis without considering IEMs
-Failing to consider dialysis for refractory hyperammonemia or very high ammonia levels.