Background. The rapid adoption of personalized medicine approaches in healthcare requires professionals to be able to manage the “omics revolution”. In this context, the genetics/genomics literacy of healthcare professionals should be in line with the continuous advances in this field, in order to implement its potential implications for the diagnosis, control and treatment of diseases. The present study investigates the effectiveness of a distance learning course on genetics and genomics targeted at medical doctors.Methods. In the context of a project funded by the Italian Ministry of Health, we developed a distance learning course, entitled Genetics and Genomics practice. The contents of the course were identified from a core curriculum in genetics for physicians and healthcare professionals, previously published. The course focused on genetic/genomic testing in clinical practice, pharmacogenomics and oncogenomic and was developed according to andragogical training methods (Problem-based Learning and Case-based Learning). We used a pre-test versus post-test study design to assess knowledge improvement on a set of 10 Multiple Choice Questions (MCQs). We analysed the proportion of correct answers for each question pre and post-test, as well as the mean score difference stratified by gender, age, professional status and medical discipline. Moreover, the same test was submitted to the participants eight months after the conclusion of the course (follow-up), in order to assess the retained knowledge.Results. An important number of Italian physicians (N= 1637) completed the course, most of which were primary care physicians (20.8%), public health professionals (11.5%) and specialist paediatricians (10.6%). We reported an improvement in the proportion of correct answers for all of the 10 MCQs set at the post-test. The overall mean score to the questions significantly increased in the post-test, from 59.46 in the pre-test to 71.42 in the post-test (p-value<0.0001). The comparison in test performance between follow-up and pre-test demonstrated an overall knowledge improvement.Conclusions. Genomic literacy among healthcare professionals is essential to ensure optimal translation to healthcare delivery of research. The results of this course suggest that distance-learning training in genetic/genomic practices represents an effective method to improve physicians’ knowledge in the immediate and mid-term time scale.