Hematopoietic cell transplantation for Diamond Blackfan anemia: A report from the Pediatric Group of the Brazilian Bone Marrow Transplantation Society

Eur J Haematol. 2020 Oct;105(4):426-433. doi: 10.1111/ejh.13463. Epub 2020 Jul 2.

Abstract

Objectives: The aim of this study was to analyze the outcomes of children with Diamond-Blackfan anemia (DBA) treated in Brazil with hematopoietic cell transplantation (HCT).

Methods: We performed a retrospective analysis of 44 pediatrics patients transplanted between 1990 and 2018. The median age of patients was 5 years, and 57% were male. Twenty-five received their first HCT from an HLA-matched sibling donor (MSD), 12 from a HLA matched unrelated bone marrow donor (MUD 10/10, n = 12) and 7 other HLA mismatched donors (MMD).

Results: After a median follow-up of 4 years, estimate 5-year overall survival (OS) for the entire cohort was 70%, 80% for MSD group, 73% for MUD, and 29% for MMD. Thirty-eight out of the 44 evaluable patients engrafted successfully. Primary and secondary graft failure was observed in five and three patients, respectively. Rates of grade II-IV and III-IV acute graft-versus-host disease (aGVHD) were 25% and 18%, respectively. Nine patients developed chronic GVHD (cGVHD).

Conclusion: Overall survival rates observed after HLA matched donors transplant for DBA were comparable to those reported from higher-income countries and international registries.

Keywords: Diamond Blackfan anemia; bone marrow transplantation; hematopoietic stem cell transplantation; matched sibling donor and matched unrelated donor.

MeSH terms

  • Anemia, Diamond-Blackfan / diagnosis
  • Anemia, Diamond-Blackfan / epidemiology
  • Anemia, Diamond-Blackfan / therapy*
  • Bone Marrow Transplantation
  • Brazil / epidemiology
  • Child
  • Child, Preschool
  • Disease Management
  • Female
  • Graft vs Host Disease / diagnosis
  • Graft vs Host Disease / etiology
  • HLA Antigens / genetics
  • Health Care Surveys
  • Hematopoietic Stem Cell Transplantation* / adverse effects
  • Hematopoietic Stem Cell Transplantation* / methods
  • Histocompatibility Testing
  • Humans
  • Infant
  • Male
  • Retrospective Studies
  • Severity of Illness Index
  • Siblings
  • Unrelated Donors

Substances

  • HLA Antigens