Genetic factors have been shown to influence occurrence and severity of several psychiatric disorders and also to modulate outcome to drug treatment. Obsessive-compulsive disorder (OCD) is a severe psychiatric condition with clear genetic roots; there is also some evidence to suggest that genetic factors may impact on response to drug treatment. Typically between 40 and 60% of patients are deemed nonresponders to antidepressant medication and clinical factors have only been modestly correlated with treatment response. Thus, identification of biological factors which may relate to treatment response could be extremely valuable in improving clinical outcome. In this article, we briefly review previous work regarding clinical and demographical factors associated with drug response in OCD, then focus on recent findings regarding candidate genes which may influence drug response, including those in the serotonin system, brain-derived neurotrophic factor and the glutamate transporter gene. The cytochrome system may also be highly relevant to drug response. Thus far, relatively few studies regarding the pharmacogenetics of OCD have been published, and therefore further investigation with functional analyses and consideration of environmental factors are warranted to facilitate clinical use of pharmacogenetic findings in the future.