Blood Res.  2017 Jun;52(2):119-124. 10.5045/br.2017.52.2.119.

Treatment and response of autoimmune cytopenia occurring after allogeneic hematopoietic cell transplantation in children

Affiliations
  • 1Division of Hematology and Oncology, Department of Pediatrics, The Catholic University of Korea, Seoul, Korea. chobinkr@catholic.ac.kr

Abstract

BACKGROUND
Autoimmune cytopenia (AIC) is a rare complication of allogeneic hematopoietic cell transplantation (HCT). In this study, we reviewed the diagnosis, treatment and response to therapy for pediatric patients with post-HCT AIC at our institution.
METHODS
Of the 292 allogeneic HCTs performed from January, 2011 to December, 2015 at the Department of Pediatrics, The Catholic University of Korea, seven were complicated by post-HCT AIC, resulting in an incidence of 2.4%.
RESULTS
All seven patients with post-HCT AIC had received unrelated donor transplant. Six of seven patients had a major donor-recipient blood type mismatch. The subtypes of AIC were as follows: immune thrombocytopenia (ITP) 2, autoimmune hemolytic anemia (AIHA) 2, Evans syndrome 3. Median time from HCT to AIC diagnosis was 3.6 months. All but one patient responded to first line therapy of steroid±intravenous immunoglobulin (IVIG), but none achieved complete response (CR) with this treatment. After a median duration of treatment of 15.3 months, two patients with ITP achieved CR and five had partial response (PR) of AIC. Five patients were treated with rituximab, resulting in the following response: 2 CR, 2 PR, 1 no response (NR). Median time to response to rituximab was 26 days from first infusion. All patients are alive without event.
CONCLUSION
Post-HCT AIC is a rare complication that may not resolve despite prolonged therapy. Rapid initiation of second line agents including but not limited to B cell depleting treatment should be considered for those that fail to achieve CR with first line therapy.

Keyword

Autoimmune cytopenia; Autoimmune hemolytic anemia; Immune thrombocytopenia; Hematopoietic cell transplantation; Rituximab

MeSH Terms

Anemia, Hemolytic, Autoimmune
Cell Transplantation*
Child*
Diagnosis
Humans
Immunoglobulins
Incidence
Korea
Pediatrics
Purpura, Thrombocytopenic, Idiopathic
Rituximab
Transplants*
Unrelated Donors
Immunoglobulins
Rituximab

Reference

1. Daikeler T, Tyndall A. Autoimmunity following haematopoietic stem-cell transplantation. Best Pract Res Clin Haematol. 2007; 20:349–360. PMID: 17448966.
Article
2. O'Brien TA, Eastlund T, Peters C, et al. Autoimmune haemolytic anaemia complicating haematopoietic cell transplantation in paediatric patients: high incidence and significant mortality in unrelated donor transplants for non-malignant diseases. Br J Haematol. 2004; 127:67–75. PMID: 15384979.
3. Faraci M, Zecca M, Pillon M, et al. Autoimmune hematological diseases after allogeneic hematopoietic stem cell transplantation in children: an Italian multicenter experience. Biol Blood Marrow Transplant. 2014; 20:272–278. PMID: 24274983.
Article
4. Chang TY, Jaing TH, Wen YC, Huang IA, Chen SH, Tsay PK. Risk factor analysis of autoimmune hemolytic anemia after allogeneic hematopoietic stem cell transplantation in children. Medicine (Baltimore). 2016; 95:e5396. PMID: 27861376.
Article
5. Page KM, Mendizabal AM, Prasad VK, 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; 14:1108–1117. PMID: 18804040.
Article
6. Wang M, Wang W, Abeywardane A, et al. Autoimmune hemolytic anemia after allogeneic hematopoietic stem cell transplantation: analysis of 533 adult patients who underwent transplantation at King's College Hospital. Biol Blood Marrow Transplant. 2015; 21:60–66. PMID: 25262883.
Article
7. Sanz J, Arriaga F, Montesinos P, et al. Autoimmune hemolytic anemia following allogeneic hematopoietic stem cell transplantation in adult patients. Bone Marrow Transplant. 2007; 39:555–561. PMID: 17351645.
Article
8. Sanz J, Arango M, Carpio N, et al. Autoimmune cytopenias after umbilical cord blood transplantation in adults with hematological malignancies: a single-center experience. Bone Marrow Transplant. 2014; 49:1084–1088. PMID: 24887383.
Article
9. Raj K, Narayanan S, Augustson B, et al. Rituximab is effective in the management of refractory autoimmune cytopenias occurring after allogeneic stem cell transplantation. Bone Marrow Transplant. 2005; 35:299–301. PMID: 15568036.
Article
10. Ship A, May W, Lucas K. Anti-CD20 monoclonal antibody therapy for autoimmune hemolytic anemia following T cell-depleted, haplo-identical stem cell transplantation. Bone Marrow Transplant. 2002; 29:365–366. PMID: 11896436.
Article
11. Hongeng S, Tardtong P, Worapongpaiboon S, Ungkanont A, Jootar S. Successful treatment of refractory autoimmune haemolytic anaemia in a post-unrelated bone marrow transplant paediatric patient with rituximab. Bone Marrow Transplant. 2002; 29:871–872. PMID: 12058238.
Article
12. Corti P, Bonanomi S, Vallinoto C, et al. Rituximab for immune hemolytic anemia following T- and B-Cell-depleted hematopoietic stem cell transplantation. Acta Haematol. 2003; 109:43–45. PMID: 12486323.
Article
13. Radhi M, Rumelhart S, Tatman D, Goldman F. Severe autoimmune hemolytic anemia after unrelated umbilical cord blood transplant for familial hemophagocytic lymphohistiocytosis: significant improvement after treatment with rituximab. J Pediatr Hematol Oncol. 2007; 29:125–127. PMID: 17279011.
Article
14. Chung NG, Lee JW, Jang PS, Jeong DC, Cho B, Kim HK. Reduced dose cyclophosphamide, fludarabine and antithymocyte globulin for sibling and unrelated transplant of children with severe and very severe aplastic anemia. Pediatr Transplant. 2013; 17:387–393. PMID: 23551397.
Article
15. Lee JW, Kim SK, Jang PS, et al. Impact of CD34+ cell dose in children who receive unrelated PBSCT with in vivo T-cell depletion for hematologic malignancies. Bone Marrow Transplant. 2015; 50:68–73. PMID: 25265463.
Article
16. Yum SK, Choi HY, Lee JW, et al. Evaluation of risk for graft-versushost disease in children who receive less than the full doses of mini-dose methotrexate for graft-versus-host disease prophylaxis in allogeneic hematopoietic stem cell transplantation. Korean J Pediatr. 2013; 56:490–495. PMID: 24348662.
Article
17. Rodeghiero F, Stasi R, Gernsheimer T, et al. Standardization of terminology, definitions and outcome criteria in immune thrombocytopenic purpura of adults and children: report from an international working group. Blood. 2009; 113:2386–2393. PMID: 19005182.
Article
18. Kotb R, Pinganaud C, Trichet C, et al. Efficacy of mycophenolate mofetil in adult refractory auto-immune cytopenias: a single center preliminary study. Eur J Haematol. 2005; 75:60–64. PMID: 15946312.
Article
19. Loh Y, Oyama Y, Statkute L, et al. Development of a secondary autoimmune disorder after hematopoietic stem cell transplantation for autoimmune diseases: role of conditioning regimen used. Blood. 2007; 109:2643–2648. PMID: 17119125.
Article
20. Quartier P, Brethon B, Philippet P, Landman-Parker J, Le Deist F, Fischer A. Treatment of childhood autoimmune haemolytic anaemia with rituximab. Lancet. 2001; 358:1511–1513. PMID: 11705566.
Article
21. Rao VK, Dugan F, Dale JK, et al. Use of mycophenolate mofetil for chronic, refractory immune cytopenias in children with autoimmune lymphoproliferative syndrome. Br J Haematol. 2005; 129:534–538. PMID: 15877736.
22. Urban C, Benesch M, Sovinz P, Schwinger W, Lackner H. Fatal Evans' syndrome after matched unrelated donor transplantation for hyper-IgM syndrome. Eur J Haematol. 2004; 72:444–447. PMID: 15128425.
Article
23. Beck JC, Burke MJ, Tolar J. Response of refractory immune thrombocytopenia after bone marrow transplantation to romiplostim. Pediatr Blood Cancer. 2010; 54:490–491. PMID: 19908296.
Article
24. Park JA, Lee HH, Kwon HS, Baik CR, Song SA, Lee JN. Sirolimus for refractory autoimmune hemolytic anemia after allogeneic hematopoietic stem cell transplantation: a case report and literature review of the treatment of post-transplant autoimmune hemolytic anemia. Transfus Med Rev. 2016; 30:6–14. PMID: 26481836.
Article
25. Cooper N, Stasi R, Cunningham-Rundles S, et al. The efficacy and safety of B-cell depletion with anti-CD20 monoclonal antibody in adults with chronic immune thrombocytopenic purpura. Br J Haematol. 2004; 125:232–239. PMID: 15059147.
Article
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