European Journal of Cancer
Volume 41, Issue 17 , Pages 2682-2689 , November 2005

Multi-centre pilot study of 2-chlorodeoxyadenosine and cytosine arabinoside combined chemotherapy in refractory Langerhans cell histiocytosis with haematological dysfunction

  • F. Bernard

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, Hôpital Arnaud de Villeneuve, CHU de Montpellier, 34000 Montpellier, France
    • Corresponding Author InformationCorresponding author. Tel.: +33 4 67 33 66 03.
  • ,
  • C. Thomas

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, Hôtel Dieu, CHU de Nantes, Quai Montcousu, 44000 Nantes, France
  • ,
  • Y. Bertrand

      Affiliations

    • Service d’Hématologie Pédiatrique, Hôpital Debrousse, 29 rue Sœur Bouvier, Lyon, France
  • ,
  • M. Munzer

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, CHU de Reims, France
  • ,
  • J. Landman Parker

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, Hôpital Trousseau, 75012, France
  • ,
  • M. Ouache

      Affiliations

    • Unité d’Immuno-Hématologie, Département de Pédiatrie, Hôpital Necker 75015 Paris, France
  • ,
  • V. Minard Colin

      Affiliations

    • Unité d’Immuno-Hématologie, Département de Pédiatrie, Hôpital Necker 75015 Paris, France
  • ,
  • Y. Perel

      Affiliations

    • Service de Médecine Infantile, CHU Pellegrin, Bordeaux, France
  • ,
  • P. Chastagner

      Affiliations

    • Service de Médecine Infantile II, Hopitaux de Brabois, Vandoeuvre les Nancy Cedex, France
  • ,
  • C. Vermylen

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, CHU Bruxelles, Belgium
  • ,
  • J. Donadieu

      Affiliations

    • Service d’Hémato-Oncologie Pédiatrique, Hôpital Trousseau, 75012, France

Received 30 December 2003 ,Revised 2 November 2004 ,Accepted 7 February 2005.

References 

  1. Gadner H, Grois N, Arico M, et al. A randomized trial of treatment for multisystem langerhans’cell histiocytosis. J Pediatr. 2001;138:728–734
  2. Minkov M, Grois N, Heitger A, et al. Response to initial treatment of multisystem Langerhans cell histiocytosis: an important prognostic indicator. Med Pediatr Oncol. 2002;39:581–585
  3. The French Langerhans’ Cell Histiocytosis Study Group. A multicentre retrospective survey of Langerhans’ cell histiocytosis: 348 cases observed between 1983 and 1993. Arch Dis Child 1996, 75, 17–24.
  4. Minkov M, Grois N, Heitger A, et al. Treatment of multisystem Langerhans cell histiocytosis. Results of the DAL-HX 83 and DAL-HX 90 studies. DAL-HX Study Group. Klin Padiatr. 2000;212:139–144
  5. Saven A, Piro LD. 2-Chlorodeoxyadenosine: a potent antimetabolite with major activity in the treatment of indolent lymphoproliferative disorders. Hematol Cell Ther. 1996;38(suppl. 2):93–101
  6. Saven A, Burian C. Cladribine activity in adult langerhans-cell histiocytosis. Blood. 1999;93:4125–4130
  7. Weitzman S, Wayne AS, Arceci R, et al. Nucleoside analogues in the therapy of Langerhans cell histiocytosis: a survey of members of the histiocyte society and review of the literature. Med Pediatr Oncol. 1999;33:476–481
  8. Dimopoulos MA, Theodorakis M, Kostis E, et al. Treatment of Langerhans cell histiocytosis with 2-chlorodeoxyadenosine. Leuk Lymphoma. 1997;25:187–189
  9. Frost JD, Wiersma SR. Progressive Langerhans cell histiocytosis in an infant with Klinefelter syndrome successfully treated with allogeneic bone marrow transplantation. J Pediatr Hematol Oncol. 1996;18:396–400
  10. Grau J, Ribera JM, Tormo M, et al. Results of treatment with 2-chlorodeoxyadenosine in refractory or relapsed Langerhans cell histiocytosis. Study of 9 patients. Med Clin (Barc). 2001;116:339–342
  11. Stine KC, Saylors RL, Williams LL, et al. 2-Chlorodeoxyadenosine (2-CDA) for the treatment of refractory or recurrent Langerhans cell histiocytosis (LCH) in pediatric patients. Med Pediatr Oncol. 1997;29:288–292
  12. Stine KC, Saylors RL, Saccente S, et al. Efficacy of continuous infusion 2-CDA (cladribine) in pediatric patients with Langerhans cell histiocytosis. Pediatr Blood Cancer. 2004;43:81–84
  13. Egeler RM, de Kraker J, Voute PA. Cytosine-arabinoside, vincristine, and prednisolone in the treatment of children with disseminated Langerhans cell histiocytosis with organ dysfunction: experience at a single institution. Med Pediatr Oncol. 1993;21:265–270
  14. Crews KR, Gandhi V, Srivastava DK, et al. Interim comparison of a continuous infusion versus a short daily infusion of cytarabine given in combination with cladribine for pediatric acute myeloid leukemia. J Clin Oncol. 2002;20:4217–4224
  15. Kornblau SM, Gandhi V, Andreef HM, et al. Clinical and laboratory studies of 2-chlorodeoxyadenosine cytosine arabinoside for relapsed or refractory acute myelogenous leukemia in adults. Leukemia. 1996;10:1563–1569
  16. Choi SW, Bangaru BS, Wu CD, et al. Gastrointestinal involvement in disseminated Langerhans cell histiocytosis (LCH) with durable complete response to 2-chlorodeoxyadenosine and high-dose cytarabine. J Pediatr Hematol Oncol. 2003;25:503–506
  17. Nagarajan R, Neglia J, Ramsay N, et al. Successful treatment of refractory Langerhans cell histiocytosis with unrelated cord blood transplantation. J Pediatr Hematol Oncol. 2001;23:629–632
  18. Histiocyte Society Writing Group Histiocytosis syndrome in children. Lancet 1987, 15, 208–209.
  19. Donadieu J, Piguet C, Bernard F, et al. A new clinical score for disease activity in Langerhans cell histiocytosis. Pediatr Blood Cancer. 2004;43:1–7
  20. Schemper M, Smith TL. A note on quantifying follow-up in studies of failure time. Control Clin Trials. 1996;17:343–346
  21. Arico M, Colella R, Conter V, et al. Cyclosporine therapy for refractory Langerhans cell histiocytosis. Med Pediatr Oncol. 1995;25:12–16
  22. Jakobson AM, Kreuger A, Hagberg H, et al. Treatment of Langerhans cell histiocytosis with alpha-interferon. Lancet. 1987;2:1520–1521
  23. Bertolini F, Mingrone W, Alietti A, et al. Thalidomide in multiple myeloma, myelodysplastic syndromes and histiocytosis. Analysis of clinical results and of surrogate angiogenesis markers. Ann Oncol. 2001;12:987–990
  24. Henter JI, Karlen J, Calming U, et al. Successful treatment of Langerhans’-cell histiocytosis with etanercept. N Engl J Med. 2001;345:1577–1578
  25. McCowage GB, Frush DP, Kurtzberg J. Successful treatment of two children with Langerhans’ cell histiocytosis with 2′-deoxycoformycin. J Pediatr Hematol Oncol. 1996;18:154–158
  26. Akkari V, Donadieu J, Piguet C, et al. Hematopoietic stem cell transplantation in patients with severe Langerhans cell histiocytosis and hematological dysfunction: experience of the French Langerhans Cell Study Group. Bone Marrow Transpl. 2003;31:1097–1103
  27. Betticher DC, Fey MF, von Rohr A, et al. High incidence of infections after 2-chlorodeoxyadenosine (2-CDA) therapy in patients with malignant lymphomas and chronic and acute leukaemias. Ann Oncol. 1994;5:57–64
  28. Chang SE, Koh GJ, Choi JH, et al. Widespread skin-limited adult Langerhans cell histiocytosis: long-term follow-up with good response to interferon alpha. Clin Exp Dermatol. 2002;27:135–137
  29. Conias S, Strutton G, Stephenson G. Adult cutaneous Langerhans cell histiocytosis. Australas J Dermatol. 1998;39:106–108
  30. Dallafior S, Pugin P, Cerny T, et al. Successful treatment of a case of cutaneous Langerhans cell granulomatosis with 2-chlorodeoxyadenosine and thalidomide. Hautarzt. 1995;46:553–560
  31. Goh NS, McDonald CE, MacGregor DP, et al. Successful treatment of Langerhans cell histiocytosis with 2-chlorodeoxyadenosine. Respirology. 2003;8:91–94
  32. Ottaviano F, Finlay JL. Diabetes insipidus and Langerhans cell histiocytosis: a case report of reversibility with 2-chlorodeoxyadenosine. J Pediatr Hematol Oncol. 2003;25:575–577
  33. Pardanani A, Phyliky RL, Li CY, et al. 2-Chlorodeoxyadenosine therapy for disseminated Langerhans cell histiocytosis. Mayo Clin Proc. 2003;78:301–306
  34. Robak T, Kordek R, Robak E, et al. Langerhans cell histiocytosis in a patient with systemic lupus erythematosus: a clonal disease responding to treatment with cladribine, and cyclophosphamide. Leuk Lymphoma. 2002;43:2041–2046
  35. Rodriguez-Galindo C, Kelly P, Jeng M, et al. Treatment of children with Langerhans cell histiocytosis with 2-chlorodeoxyadenosine. Am J Hematol. 2002;69:179–184
  36. Saven A, Figueroa ML, Piro LD, et al. 2-Chlorodeoxyadenosine to treat refractory histiocytosis X. N Engl J Med. 1993;329:734–735
  37. Saven A, Foon KA, Piro LD. 2-Chlorodeoxyadenosine-induced complete remissions in Langerhans-cell histiocytosis. Ann Int Med. 1994;121:430–432
  38. Watts J, Files B. Langerhans cell histiocytosis: central nervous system involvement treated successfully with 2-chlorodeoxyadenosine. Pediatr Hematol Oncol. 2001;18:199–204
  39. Gandhi V, Estey E, Keating MJ, et al. Chlorodeoxyadenosine and arabinosylcytosine in patients with acute myelogenous leukemia: pharmacokinetic, pharmacodynamic, and molecular interactions. Blood. 1996;87:256–264
  40. Chasty RC, Myint H, Oscier DG, et al. Autoimmune haemolysis in patients with B-CLL treated with chlorodeoxyadenosine (CDA). Leuk Lymphoma. 1998;29:391–398

PII: S0959-8049(05)00154-1

doi: 10.1016/j.ejca.2005.02.007

European Journal of Cancer
Volume 41, Issue 17 , Pages 2682-2689 , November 2005