Drug response is known to be highly variable among treated patients and affected by many factors, such as age, sex, ethnicity, concomitant diseases, and pharmacological therapy. However, sequence variants in the human genome are now considered an important cause of differences in drug responses. Pharmacogenetics, which is the utilization of individual genetic data to predict the outcome of drug treatment with respect to both beneficial and adverse effects, represents an emerging field of genetics with the potential to become useful for the identification of the most effective drug and the most beneficial dose for a given individual. On the basis of these considerations and thanks to recent advances in genetics and molecular biology, pharmacogenetics is becoming a flowering field in both basic and clinical research. Nevertheless, to date the opportunity to apply pharmacogenetic approaches to drug response and the possibility to use genetic screenings to tailor decisions about pharmacological treatments have limited applications. And this is even truer in the field of osteoporosis, in which pharmacogenetic studies are in their infancy. In this paper we review the most recent data on pharmacogenetics of osteoporosis, highlighting the presentations at the Second International Meeting on Pharmacogenetics of Osteoarticular Disorders held in Florence in April 2008.