ABSTRACT Introduction: In Africa, where access to diagnosis and treatment of hemophilia is the lowest in the world, prophylaxis is rarely used in preference to on-demand treatment. There are limited data of prophylaxis treatment from sub-Saharan Africa. The aim of this study was to evaluate clinical outcomes and inhibitor development in people with hemophilia receiving low-dose prophylaxis (LDP) in a sub-Saharan African setting. Methods: We conducted a three-year prospective study. A once or twice weekly prophylaxis regimen of 25 IU/kg of rFVIIIFc or 30 IU/kg of rFIXFc was given to Hemophilia A and B, respectively. We evaluated clinical outcomes and inhibitors occurrence, determined by screening and titration using the Nijmegen technique. Results: A total of 15 patients were included in the LDP regimen. The mean age was 6.3 years (1.5 - 10). A significant reduction was noted in the annualized bleeding rate, from 7.53 to 1.33 (p = 0.0001); the annualized joint bleeding rate passed from 3.6 to 1.4 (p = 0.001) and the proportion of severe bleeding, from 86.1% to 16.7% (p = 0.0001). The Hemophilia Joint Health Score (HJHS) moved from 9.6 to 3.4 (p = 0.0001) and the Functional Independence Score in Hemophilia (FISH) improved from 25.8 to 30.9 (p = 0.0001). School absenteeism decreased from 7.33% to 2.59%. Adherence to prophylaxis was 89.5% versus 60%. Consumption was 580 IU/kg/year versus 1254.6 IU/kg/year before and after prophylaxis, respectively. Incidence of inhibitors was 23% (3 /13 HA). Conclusion: The LDP in Hemophilia improves the clinical outcome without a surplus risk of inhibitor development. Using extended half-life clotting factor concentrates (CFCs) is better for prophylaxis in resource-limited countries, as they allow better compliance in treatment. Introduction Africa world ondemand on demand subSaharan sub Saharan lowdose low dose (LDP setting Methods threeyear three year 2 IUkg IU kg 3 B respectively occurrence technique Results 1 63 6 6. 1.5 5 (1. 10. 10 . 10) 753 7 53 7.5 133 33 1.3 p 0.0001 00001 0 0001 36 3. 14 4 1. 0.001 001 861 86 86.1 167 16 16.7 0.0001. HJHS (HJHS 96 9 9. 34 FISH (FISH 258 8 25. 309 30. 733 7.33 259 59 2.59% 895 89 89.5 60 60% 58 IUkgyear 12546 1254 1254. 23 ( 13 /1 HA. HA HA) Conclusion halflife half life CFCs (CFCs resourcelimited resource countries (1 75 7. 0.000 0000 000 0.00 00 86. 16. 73 7.3 2.59 89. 125 / 0.0 2.5 12 0. 2.