Nonmyeloablative allogeneic hematopoietic stem cell transplantation for GATA2 deficiency

Biol Blood Marrow Transplant. 2014 Dec;20(12):1940-8. doi: 10.1016/j.bbmt.2014.08.004. Epub 2014 Aug 9.

Abstract

We treated 14 patients with GATA2 deficiency using a nonmyeloablative allogeneic hematopoietic stem cell transplantation regimen. Four patients received peripheral blood stem cells from matched related donors (MRD), 4 patients received peripheral blood stem cells from matched unrelated donors (URD), 4 patients received hematopoietic stem cells from umbilical cord blood donors (UCB), and 2 patients received bone marrow cells from haploidentical related donors. MRD and URD recipients received conditioning with 3 days of fludarabine and 200 cGy total body irradiation (TBI). Haploidentical related donor recipients and UCB recipients received cyclophosphamide and 2 additional days of fludarabine along with 200 cGY TBI. MRD, URD, and UCB recipients received tacrolimus and sirolimus for post-transplantation immunosuppression, whereas haploidentical recipients received high-dose cyclophosphamide followed by tacrolimus and mycophenolate mofetil. Eight patients are alive with reconstitution of the severely deficient monocyte, B cell, and natural killer cell populations and reversal of the clinical phenotype at a median follow-up of 3.5 years. Two patients (1 URD recipient and 1 UCB recipient) rejected the donor graft and 1 MRD recipient relapsed with myelodysplastic syndrome after transplantation. We are currently using a high-dose conditioning regimen with busulfan and fludarabine in patients with GATA2 deficiency to achieve more consistent engraftment and eradication of the malignant myeloid clones.

Keywords: Familial acute myelogenous leukemia; GATA2; Hematopoietic stem cell transplantation; Myelodysplastic syndrome.

Publication types

  • Clinical Trial
  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Adolescent
  • Adult
  • Allografts
  • Antineoplastic Agents / administration & dosage
  • Bone Marrow Transplantation*
  • Child
  • Cord Blood Stem Cell Transplantation*
  • Female
  • Follow-Up Studies
  • GATA2 Transcription Factor / deficiency*
  • Genetic Diseases, Inborn / blood
  • Genetic Diseases, Inborn / genetics
  • Genetic Diseases, Inborn / therapy*
  • Hematopoietic Stem Cell Transplantation*
  • Humans
  • Immunologic Deficiency Syndromes / genetics
  • Immunologic Deficiency Syndromes / therapy*
  • Immunosuppressive Agents / administration & dosage
  • Male
  • Middle Aged
  • Sirolimus / administration & dosage
  • Tacrolimus / administration & dosage
  • Transplantation Conditioning*
  • Unrelated Donors
  • Vidarabine / administration & dosage
  • Vidarabine / analogs & derivatives
  • Whole-Body Irradiation

Substances

  • Antineoplastic Agents
  • GATA2 Transcription Factor
  • GATA2 protein, human
  • Immunosuppressive Agents
  • Vidarabine
  • fludarabine
  • Sirolimus
  • Tacrolimus