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 Blood, 1 February 2006, Vol. 107, No. 3, pp. 1233-1236.
Prepublished online as a Blood First Edition Paper on October 11, 2005; DOI 10.1182/blood-2005-05-1819.

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TRANSPLANTATION
Brief report

Stem cell transplantation with reduced-intensity conditioning for hemophagocytic lymphohistiocytosis

Nichola Cooper, Kanchan Rao, Kimberly Gilmour, Lema Hadad, Stuart Adams, Cathy Cale, Graham Davies, David Webb, Paul Veys, and Persis Amrolia

From the Great Ormond Street Hospital for Children NHS Trust, London, United Kingdom.

Allogeneic stem cell transplantation (SCT) is curative for hemophagocytic lymphohistiocytosis (HLH). However, patients frequently have significant morbidity before transplantation and there is high transplant-related mortality (TRM). Because first-degree HLH is caused by immune dysregulation, a reduced-intensity conditioned (RIC) regimen might be sufficient for cure while decreasing the TRM. Twelve patients with HLH underwent RIC SCT from a matched family/unrelated or haploidentical donor. Eleven were conditioned with fludarabine/melphalan with additional busulphan for haploidentical grafts. One received fludarabine and 2-Gy total body irradiation (TBI). All patients showed engraftment at a median of 14 days. Nine of 12 (75%) are alive and in complete remission (CR) a median of 30 months (range, 9-73 months) after SCT. Two patients died from pneumonitis and one from hepatic rupture. Four patients developed acute graft-versus-host disease (GVHD) and 3 have chronic GVHD. Three of 9 survivors have mixed chimerism but remain free of disease. In summary, RIC compares favorably to conventional SCT with long-term disease control in surviving patients despite a significant incidence of mixed chimerism.


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