OBJECTIVES: Relapse following allogenic hematopoietic stem cell transplant (aHSCT) is usually associated with poor outcomes. The impact of treatment choice on costs and survival remains unknown. The objective of this study was to measure the economic burden associated with the management of relapse following aHSCT and the impact of treatment on survival and healthcare costs. METHODS: A retrospective medical chart review was conducted in a HSCT specialized center in Montreal (Canada). Eligible patients were diagnosed with acute leukemia (AL) or myelodysplastic syndrome (MDS) and relapsed following a human leukocyte antigen (HLA) identical aHSCT between January 2011 and December 2014. Patients and disease characteristics as well as relapse-related healthcare resources were collected from date of relapse until death or last follow-up. Canadian unit costs for each resource were obtained from literature and governmental publications. RESULTS: A total of 36 patients were included in the analysis. Of these, 4 were diagnosed with MDS and 32 with AL. Treatment approaches following aHSCT relapse were divided in three groups: group 1 received supportive care (n=9), group 2 received chemotherapy or tyrosine kinase inhibitors (n=21) and group 3 received a cellular based therapy, either donor lymphocyte infusion or a second aHSCT (n=6). The mean cost per patient per month was C$20,239 (SD=17,079). The mean survival following relapse was 12.4 months (SD=2.8). For group 1, 2 and 3, the mean cost per patient per month was C$17,436 (SD=16,447), C$22,914 (SD=18,474), C$15,082 (SD=12,954), respectively. For group 1, 2 and 3, the mean survival was 4.0 months (SD=2.0), 7.2 months (SD=1.6), and 44.6 months (SD=8.4), respectively. CONCLUSIONS: Results demonstrate that compared to other treatments, cellular therapy following aHSCT is associated with a prolonged survival at a similar cost per patient per month and appear cost effective. Further studies with larger sample sizes are needed to confirm these findings.