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Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation.

Publication ,  Journal Article
Page, KM; Mendizabal, AM; Prasad, VK; Martin, PL; Parikh, S; Wood, S; Sempowski, GD; Szabolcs, P; Kurtzberg, J
Published in: Biol Blood Marrow Transplant
October 2008

Autoimmune cytopenias are a recognized complication of hematopoietic stem cell transplant (HSCT), and are considered to be a feature of chronic graft-versus-host disease (cGVHD). We report on a cohort of very young infants (< or =3 months of age) receiving HSCT from unrelated donor umbilical cord blood for genetic disorders who developed posttransplant autoimmune cytopenias at an increased rate compared to older aged controls. These infants received a conditioning regimen consisting of busulfan, cyclophosphamide, and antithymocyte globulin (ATG). All infants received HLA mismatched unrelated umbilical cord blood as graft source. GVHD prophylaxis was either cyclosporine + methylprednisolone (n = 16) or cyclosporine + mycophenolate mofetil (n = 3). Engraftment, acute GVHD (aGVHD) and cGVHD, survival, treatment-related mortality (TRM), and deaths were evaluated. Ten patients developed cGVHD manifesting as autoimmune cytopenias at a median 247 days posttransplant with a cumulative incidence of 44% (95% confidence interval [CI] 21%-68%) and 56% (95% CI 32%-80%) at 1 and 2 years, respectively. In 6 of 10 patients developing autoimmune cytopenias, cGVHD presented as autoimmune cytopenia de novo. The cytopenias observed included anemia (n = 4), thrombocytopenia (n = 1), anemia with thrombocytopenia (n = 3), and pancytopenia (n = 2). No graft factors were identified as being significant to development of cGVHD. All patients responded to treatment with methylprednisolone, azithioprine +/- rituximab. One patient required splenectomy. We hypothesize that posttransplant immunosuppression interferes with normal immune ontogeny creating immune dysregulation and graft directed cell destruction. Alternative strategies to prevent GVHD should be considered for this unique patient population.

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Published In

Biol Blood Marrow Transplant

DOI

EISSN

1523-6536

Publication Date

October 2008

Volume

14

Issue

10

Start / End Page

1108 / 1117

Location

United States

Related Subject Headings

  • Transplantation Conditioning
  • Thrombocytopenia
  • Pancytopenia
  • Lysosomal Storage Diseases
  • Infant, Newborn
  • Infant
  • Immunology
  • Humans
  • Histocompatibility
  • Hemoglobinopathies
 

Citation

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Page, K. M., Mendizabal, A. M., Prasad, V. K., Martin, P. L., Parikh, S., Wood, S., … Kurtzberg, J. (2008). Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation. Biol Blood Marrow Transplant, 14(10), 1108–1117. https://doi.org/10.1016/j.bbmt.2008.07.006
Page, Kristin M., Adam M. Mendizabal, Vinod K. Prasad, Paul L. Martin, Suhag Parikh, Susan Wood, Gregory D. Sempowski, Paul Szabolcs, and Joanne Kurtzberg. “Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation.Biol Blood Marrow Transplant 14, no. 10 (October 2008): 1108–17. https://doi.org/10.1016/j.bbmt.2008.07.006.
Page KM, Mendizabal AM, Prasad VK, Martin PL, Parikh S, Wood S, et al. Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation. Biol Blood Marrow Transplant. 2008 Oct;14(10):1108–17.
Page, Kristin M., et al. “Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation.Biol Blood Marrow Transplant, vol. 14, no. 10, Oct. 2008, pp. 1108–17. Pubmed, doi:10.1016/j.bbmt.2008.07.006.
Page KM, Mendizabal AM, Prasad VK, Martin PL, Parikh S, Wood S, Sempowski GD, Szabolcs P, Kurtzberg J. Posttransplant autoimmune hemolytic anemia and other autoimmune cytopenias are increased in very young infants undergoing unrelated donor umbilical cord blood transplantation. Biol Blood Marrow Transplant. 2008 Oct;14(10):1108–1117.
Journal cover image

Published In

Biol Blood Marrow Transplant

DOI

EISSN

1523-6536

Publication Date

October 2008

Volume

14

Issue

10

Start / End Page

1108 / 1117

Location

United States

Related Subject Headings

  • Transplantation Conditioning
  • Thrombocytopenia
  • Pancytopenia
  • Lysosomal Storage Diseases
  • Infant, Newborn
  • Infant
  • Immunology
  • Humans
  • Histocompatibility
  • Hemoglobinopathies