ArticleBiology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation2015
Preemptive Bone Marrow Transplantation for FANCD1/BRCA2.
Article in Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed.
- Preemptive hematopoietic stem cell transplantation inHaematologica · 2026Article
- Fanconi anemia caused by biallelic inactivation of BRCA2 can present with an atypical cancer phenotype in adulthood.Clinical genetics · 2023Article
- Monitoring and treatment of MDS in genetically susceptible persons.Hematology. American Society of Hematology. Education Program · 2019Article
- Inherited bone marrow failure syndromes: considerations pre- and posttransplant.Hematology. American Society of Hematology. Education Program · 2017Review
- Review
- The Second Pediatric Blood and Marrow Transplant Consortium International Consensus Conference on Late Effects after Pediatric Hematopoietic Cell Transplantation: Defining the Unique Late Effects of Children Undergoing Hematopoietic Cell Transplantation for Immune Deficiencies, Inherited Marrow Failure Disorders, and Hemoglobinopathies.Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation · 2017Review
- Preemptive Bone Marrow Transplantation and Event-Free Survival in Fanconi Anemia.Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation · 2016Article
- Twenty years of the Italian Fanconi Anemia Registry: where we stand and what remains to be learned.Haematologica · 2016Article
- Genetic predisposition to myelodysplastic syndrome and acute myeloid leukemia in children and young adults.Leukemia & lymphoma · 2016Article
Corrections and comments
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Authors and funding
4 authors.
Funding
Abstract
Children with biallelic mutations in FANCD1/BRCA2 are at uniquely high risks of leukemia and solid tumors. Preemptive bone marrow transplantation (PE-BMT) has been proposed to avoid the development of leukemia, but empirical study of PE-BMT is unlikely because of the rarity of these children and the unknown benefit of PE-BMT. We used survival analysis to estimate the risks of leukemia and the expected survival if leukemia could be eliminated by curative PE-BMT. We used the results in a decision analysis model to explore the plausibility of PE-BMT for children with variable ages at diagnosis and risks of transplantation-related mortality. For example, PE-BMT at 1 year of age with a 10% risk of transplantation-related mortality increased the mean survival by 1.7 years. The greatest benefit was for patients diagnosed between 1 and 3 years of age, after which the benefit of PE-BMT decreased with age at diagnosis, and the risk of death from solid tumors constituted a relatively greater burden of mortality. Our methods may be used to model survival for other hematologic disorders with limited empirical data and a pressing need for clinical guidance.
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