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Immunosuppressive therapy with anti-thymocyte globulin and cyclosporine A in selected children with hypoplastic refractory cytopenia

From the Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, University of Freiburg, Germany (AY, AF, PN, CN); Institute of Pathology, Klinikum Bayreuth, Germany (IB); Department of Pediatric Hematology and Oncology, Dr von Haunersches Kinderspital, Children Hospital of the Ludwig-Maximilians-University of Munich, Munich, Germany (MF); University Children’s Hospital, University of Zürich, Switzerland (EB); Department of Pediatric Oncology, Hematology and Immunology, University of Düsseldorf, Düsseldorf, Germany (UG); Department for Pediatric Hematology/Oncology, Hannover Medical School, Hannover, Germany (K-WS); University Children’s Hospital, University of Frankfurt, Germany (TK); Department of Pediatric Hematology and Oncology, Olgaspital, Stuttgart, Germany (UG-W); Erasmus University Medical Center-Sophia Children's Hospital, Rotterdam, The Netherlands (MMvdH-E).

Correspondence: Charlotte M Niemeyer M.D., Department of Pediatrics and Adolescent Medicine, Division of Pediatric Hematology and Oncology, University of Freiburg, Mathildenstrasse 1, 79106 Freiburg, Germany. E-mail: charlotte.niemeyer{at}uniklinik-freiburg.de

It is currently unknown whether immunosuppressive therapy or hematopoietic stem cell transplantation is the most appropriate treatment strategy for children with refractory cytopenia and normal karyotype or trisomy 8. We report on 31 children with hypoplastic refractory cytopenia treated with immunosuppressive therapy consisting of antithymocyte globulin and cyclosporine. At 6 months, 22 of 29 evaluable patients had a complete or partial response; a total of ten patients achieved a complete response at varying time points. Six patients subsequently received a transplant because of non-response, progression to advanced myelodysplastic syndrome or evolution of monosomy 7. Overall and failure-free survival rates at 3 years were 88% and 57%, respectively.

Key words: myelodysplastic syndrome, refractory cytopenia, immunosuppressive therapy, anti-thymocyte globulin, children.