Risperidone is an atypical antipsychotic that has been effectively used to treat several psychiatric diseases. Atypical antipsychotics present some advantages over conventional antipsychotics, primarily because they offer effective treatment alternatives that are relatively free of extrapyramidal symptoms. However, as with all antipsychotics, there are wide individual differences in response to risperidone, both regarding therapeutic effects and adverse effects, imposing some limitations with respect to the therapeutic use of the drug. Genetic factors are thought to play an important role in determining the variability to drug response. A growing number of studies are investigating how genetic polymorphisms of enzymes involved in drug metabolism or of receptors targeted by antipsychotic agents influence drug treatment of several neuropsychiatric diseases. In this article we will review the genetic variability in both the pharmacokinetics of risperidone action and in pharmacodynamic structures mediating risperidone effects, as well as the pharmacogenetic studies performed for these genes.