Interindividual variability in the disposition and action associated with similar doses of a given medication is an inherent characteristic of both adult and pediatric populations. Genotype-phenotype relationships in infants and children must take into account the role that ontogeny plays in producing variability in both pharmacokinetics and pharmacodynamics. This review explores pharmacogenomics in the context of ontogeny and relates these to the expression of drug-metabolizing enzymes and transporters and the consequent effect on the exposure-response relationship in the early years of life.