PROJECT SUMMARY The Johns Hopkins Predoctoral Training Program in Human Genetics (HG) has long been a nationally preeminent training environment for human genetic research. It has grown steadily since its inception (1980) in parallel to the explosive growth of genetics and genomics, and their application to medicine. Although supported by preceptor faculty across 20 diverse departments, the HG has its administrative and philosophical home in the McKusick-Nathans Department of Genetic Medicine (DGM). Eighty faculty mentors engage in training our current cohort of sixty-one students. The preceptors’ research includes human and model organism genetics and genomics, study of genes and variants underlying human monogenic disorders and complex traits, quantitative genetics, gene therapy, oncogenetics, stem cells, technology development, big data and machine learning. An average of 12 students graduate each year and obtain their PhD in 5.3 years (average). The objectives of the HG program are: (1) to provide a biomedical curriculum with breadth and depth; (2) to ensure an understanding of the fundamental roles of genetic variation in human biology, diversity, and disease; (3) to provide robust training in rigorous, reproducible, responsible, and ethical research; (4) to equip our trainees with relevant professional skills and the opportunity to explore career options; and (5) to recruit and foster a diverse and inclusive training environment for all. In the first year, we introduce students to the evolving concepts of the gene, molecular biology, genomics, cell structure and dynamics, pathways and regulation, extending deeper with advanced topics in human genetics. We introduce a new computational bootcamp to provide fundamental computational and statistical skills, a theme that is iterated throughout their courses. In their second year we have developed two highly innovative courses: Systems, Genes and Mechanisms in Disease and Genomic Technologies Applications and Considerations. The courses integrate didactic presentation with student-led seminars, team-based problem solving and discussion, placing their genetic training in a human biological and pathological context and equipping them for the design and implementation of contemporary genomic experimental strategies. We provide training in rigorous and ethical experimental design and implementation, and career planning and professional development through workshops, internship opportunities and required courses. Our preceptors receive mentoring training; are appropriately mentored; and they are accountable. The overall goal of our HG program is to educate the next generation of leaders in human genetics. We seek to train independent scientists who are passionately curious about the role that genetic variation plays in the human condition. This education prepares our students to answer important basic science questions and to translate this information into bio-medical advances. The success of our g...