Workflow for the study by Reilly et al. By analyzing a cohort of 1514 MDS patients enrolled in the CIBMTR database, the authors identified rare TERT variants, quantified their functional impact on telomere elongation, and proved their clinical relevance, eventually laying the foundations for evidence-based indication for patients screening and risk-adapted protocols. CTE, C-terminal extension [domain]; NMR, nonrelapse mortality; RTD, reverse transcriptase domain; SCT, stem cell transplantation; TEN, telomerase essential N-terminal [domain]; TRBD, telomerase RNA-binding domain.