Abstract
A 32-yr-old male diagnosed with myelodysplastic syndrome underwent an unmanipulated, unrelated, HLA matched, peripheral blood stem cell transplantation. The patient and donor were both blood type O, CcDEe. Twelve weeks post-transplantation, he developed acute autoimmune hemolytic anemia (AIHA). He was transfused multiple times with washed O red cells. High-dose steroid therapy was initiated and he underwent splenectomy; however, AIHA was refractory to therapy. The patient was further treated with combined treatment modalities including immunosuppressive therapy with mycophenolate mofetil and cyclosporine and three cycles of plasma exchange, and AIHA responded to treatment. This is the third case of AIHA complicating hematopoietic stem cell transplantation reported in Korea. Since AIHA is relatively common after hematopoietic stem cell transplantation, accurate and timely diagnosis of the disease and treatment strategies with multiple modalities are necessary.
REFERENCES
1.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. 29:871–2.
2.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. 29:125–7.
3.Mullen CA., Thompson JN., Richard LA., Chan KW. Unrelated umbilical cord blood transplantation in infancy for mucopolysaccharidosis type IIB (Hunter syndrome) complicated by autoimmune hemolytic anemia. Bone Marrow Transplant. 2000. 25:1093–7.
4.Sevilla J., Gonzalez-Vicent M., Madero L., Diaz MA. Acute autoimmune hemolytic anemia following unrelated cord blood transplantation as an early manifestation of chronic graft-versus-host disease. Bone Marrow Transplant. 2001. 28:89–92.
5.Sokol RJ., Stamps R., Booker DJ., Scott FM., Laidlaw ST., Vandenberghe EA, et al. Posttransplant immune-mediated hemolysis. Transfusion. 2002. 42:198–204.
6.O'Brien TA., Eastlund T., Peters C., Neglia JP., Defor T., Ramsay NK, 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.
7.Sanz J., Arriaga F., Montesinos P., Orti G., Lorenzo I., Cantero S, et al. Autoimmune hemolytic anemia following allogeneic hematopoietic stem cell transplantation in adult patients. Bone Marrow Transplant. 2007. 39:555–61.
8.Horn B., Viele M., Mentzer W., Mogck N., DeSantes K., Cowan M. Autoimmune hemolytic anemia in patients with SCID after T cell-depleted BM and PBSC transplantation. Bone Marrow Transplant. 1999. 24:1009–13.
9.Chen FE., Owen I., Savage D., Roberts I., Apperley J., Goldman JM, et al. Late onset haemolysis and red cell autoimmunisation after allogeneic bone marrow transplant. Bone Marrow Transplant. 1997. 19:491–5.
10.Winiarski J., Ringden O., Remberger M., Dalianis T., Ljungman P., Borgstrom B. Bone marrow transplantation in children using unrelated donors at Huddinge Hospital. Acta Paediatr. 1996. 85:327–35.
11.Drobyski WR., Potluri J., Sauer D., Gottschall JL. Autoimmune hemolytic anemia following T cell-depleted allogeneic bone marrow transplantation. Bone Marrow Transplant. 1996. 17:1093–9.
12.Baek JH., Shon SK., Ahn BM., Moon JH., Jeon SB., Sung WJ, et al. Uncommon hematologic manifestations of GVHD after allogeneic stem cell transplantation: two cases with hypereosinophilia or autoimmune hemolytic anemia. Korean J Hematopoietic Stem Cell Transplant. 2004. 9:34–7. (백진호, 손상균, 안병민, 문준호, 전석봉, 성우진 등. 동종 이식 후 이식편대 숙주병의 드문 혈액학적 발현: 호산구증다증 및 자가면역성 용혈성빈혈. 대한조혈모세포이식학회지 2004;9: 34-7.).
13.Park HK., Oh SH., Choi KM., Lee HK., Kim DW. Cold agglutinin disease due to Anti-Pr cold autoantibody in a patient with bone marrow transplantation. Korean J Blood Transfusion. 2003. 14:229–33. (박현경, 오수현, 최광모, 이홍기, 김대원. 골수이식 환자에서 발생한 항-Pr 한랭 자가항체에 의한 한랭 응집소병. 대한수혈학회지 2003;14: 229-33.).
15.Brecher ME, editor. Technical manual. 15th ed.Bethesda: American Association of Blood Banks;2005. p. 629–30.
16.Leo A., Mytilineos J., Voso MT., Weber-Nordt R., Liebisch P., Lensing C, et al. Passenger lymphocyte syndrome with severe hemolytic anemia due to an anti-Jka after allogeneic PBPC transplantation. Transfusion. 2000. 40:632–6.
17.Reed M., Yearsley M., Krugh D., Kennedy MS. Severe hemolysis due to passenger lymphocyte syndrome after hematopoietic stem cell transplantation from an HLA-matched related donor. Arch Pathol Lab Med. 2003. 127:1336–8.
18.Hoegler W., Nachbaur D., Kropshofer G., Wehl G., Schoenitzer D., Nussbaumer W. Abrupt onset of severe passenger lymphocyte syndrome after HLA-mismatched unrelated bone marrow transplantation in a seven-year-old boy. Med Pediatr Oncol. 2002. 38:143–4.
19.Leo A., Lenz V., Winteroll S. Nonhemolytic passenger lymphocyte syndrome with anti-Jka after allogeneic peripheral blood progenitor cell transplantation. Transfusion. 2004. 44:1259–60.
20.Evans RS., Takahashi K., Duane RT., Payne R., Liu C. Primary thrombocytopenic purpura and acquired hemolytic anemia; evidence for a common etiology. AMA Arch Intern Med. 1951. 87:48–65.
21.Keung YK., Cobos E., Bolanos-Meade J., Issarachai S., Brideau A., Morgan D. Evans syndrome after autologous bone marrow transplant for recurrent Hodgkin's disease. Bone Marrow Transplant. 1997. 20:1099–101.
22.Kim YM., Kim BJ., Lee YH. Evans syndrome following unrelated bone marrow transplantation. Korean J Pediatr Hematol Oncol. 2003. 10:277–81. (김영명, 김봉진, 이영호. 비혈연 골수이식 후에 발생한 Evans 증후군 1예. 대한소아혈액종양학회지 2003;10: 277-81.).
23.Lee YH., Kim YD., Kim YM. Evans syndrome following T cell-repleted unrelated bone marrow transplantation for myelodysplastic syndrome: successful response to high-dose corticosteroid. Korean J Hematol. 2006. 41:204–7. (이영호, 김영대, 김영명. 고용량 부신피질호르몬에 반응을 보인 비혈연골수이식 후의 Evans 증후군. 대한혈액학회지 2006;41: 204-7.).
24.Klumpp TR., Caligiuri MA., Rabinowe SN., Soiffer RJ., Murray C., Ritz J. Autoimmune pancytopenia following allogeneic bone marrow transplantation. Bone Marrow Transplant. 1990. 6:445–7.
25.Sinha AA., Lopez MT., McDevitt HO. Autoimmune diseases: the failure of self tolerance. Science. 1990. 248:1380–8.
26.Gao EK., Loh D., Cheyney R., Kanagawa O., Sprent J. Abnormal differentiation of thymocytes in mice treated with cyclosporin A. Nature. 1988. 336:176–9.
27.McMillan MA., Muirhead CS., Lucie NP., Briggs JD., Junor BJ. Autoimmune haemolytic anaemia related to cyclosporin with ABO-compatible kidney donor and recipient. Nephrol Dial Transplant. 1991. 6:57–9.
28.Dundar S., Ozdemir O., Ozcebe O. Cyclosporin in steroid-resistant auto-immune haemolytic anaemia. Acta Haematol. 1991. 86:200–2.
29.Zecca M., Nobili B., Ramenghi U., Perrotta S., Amendola G., Rosito P, et al. Rituximab for the treatment of refractory autoimmune hemolytic anemia in children. Blood. 2003. 101:3857–61.
30.McMahon C., Babu L., Hodgson A., Hayat A., Connell NO., Smith OP. Childhood refractory autoimmune haemolytic anaemia: is there a role for anti-CD20 therapy (rituximab)? Br J Haematol. 2002. 117:480–3.
31.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. 29:365–6.
32.Corti P., Bonanomi S., Vallinoto C., Balduzzi A., Uderzo C., Cazzaniga G, et al. Rituximab for immune hemolytic anemia following T- and B-Cell-depleted hematopoietic stem cell transplantation. Acta Haematol. 2003. 109:43–5.
33.Lee JH., Lee KS. A case of autoimmune hemolytic anemia treated with rituximab in a child. Korean J Hematol. 2006. 41:321–5. (이지혜 및 이건수. Rituximab으로 자가면역용혈빈혈 치료를 시행한 소아 1예. 대한혈액학회지 2006;41: 321-5.).