Abstract:  Congenital amegakaryocytic thrombocytopenia (CAMT) is characterized by severe thrombocytopenia and the absence of megakaryocytes in bone marrow. Furthermore, mutation of the c‐mpl gene has been identified as a cause of this disorder. The only curative treatment is allogeneic stem cell transplantation (SCT). The current report describes a patient exhibiting c‐mpl mutation in both alleles who underwent transplantation of allogeneic bone marrow donated by her brother, a c‐mpl mutated carrier, employing a fludarabine‐based conditioning regimen. Engraftment and reconstitution of hematopoietic cells was rapid and without complications. These findings suggest that the carrier donor displaying the c‐mpl mutation can serve as a donor source for SCT.