Abstract: Fundamental aspects of eukaryotic molecular and cellular biology are extensively studied in the budding yeast Saccharomyces cerevisiae. Genome maintenance pathways are highly conserved and research into a number of human genetic disorders with increased genome instability and cancer predisposition have benefited greatly from studies in budding yeast. Here, we present some of the examples where yeast research into DNA damage responses and telomere maintenance pathways paved the way to understanding these processes, and their involvement in selected human diseases.